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• Tympanomastoidectomy: Comparison between canal wall-down and canal wall-up techniques in surgery<br />

for chronic otitis media<br />

• Endoscopic surgery in the treatment of crista galli pneumatization evolving with localizated frontal<br />

headaches<br />

• Velopharyngeal dysfunction: a systematic review of major instrumental and auditory-perceptual<br />

assessments<br />

• Results obtained with a low cost software-based audiometer for hearing screening<br />

• Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with<br />

cleft lip/palate<br />

• Clinical and fiberoptic endoscopic assessment of swallowing in patients with chronic obstructive<br />

pulmonary disease<br />

• Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx<br />

• Audiological outcomes of cochlear implantation in Waardenburg Syndrome<br />

• Performance analysis of ten brands of batteries for hearing aids<br />

• Contribution of audiovestibular tests to the topographic diagnosis of sudden deafness<br />

• Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy for botulinum toxin injection:<br />

an experimental study in cadaver larynges<br />

• Correlation of cephalometric and anthropometric measures with obstructive sleep apnea severity<br />

• Use of surface electromyography in phonation studies: an integrative review<br />

• Middle ear adenoma with neuroendocrine differentiation: relate of two cases and literature review<br />

• Bullous Systemic Lupus Erythematosus: Case report<br />

• Eagle’s Syndrome<br />

• Retrolabyrinthine approach for cochlear nerve preservation in neurofibromatosis type 2 and simultaneous<br />

cochlear implantation


234


ISSN 1809-9777<br />

Official Publication of the Otorhinolaryngology Foundation and<br />

Societa Oto-Rhino-Laryngologica Latina<br />

First Electronic Journal of ENT<br />

INDEXATIONS<br />

DOAJ - Diretory of Open Access Journals.<br />

FUNPEC-RP (Foundation for Scientific Research<br />

of Ribeirão Preto).<br />

Latindex - Regional Cooperative Online<br />

Information System for Scholarly Journals from<br />

Latin America, the Caribbean, Spain and<br />

Portugal.<br />

LILACS and LILACS-Express Latin-American<br />

and Caribbean Center on Health Sciencies<br />

Information.<br />

SciELO - Scientific Electronic Library Online.<br />

SCOPUS - SciVerse (Elsevier).<br />

AFFILIATION<br />

Vol. <strong>17</strong> nº 3 - Jul/Aug/September- 2013<br />

The whole edition has 4 issues published: March,<br />

June, September and December.<br />

Periodicity: threemonthly<br />

Number printed: 5,500 issues<br />

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SUPPORT<br />

EDITOR<br />

Geraldo Pereira Jotz – UFRGS – Porto Alegre – Brazil<br />

CO-EDITOR<br />

Aline Gomes Bittencourt – USP – São Paulo – Brazil<br />

ASSOCIATED EDITORS<br />

Alergy and Olfact: ................................ João Ferreira de Mello Junior ........... USP – São Paulo / Brazil<br />

Audiology: ........................................... Marcelo M. Hueb ........................... UFTM – Uberaba / Brazil<br />

Skull Base: ........................................... Ricardo L. Carrau ........................... Ohio State University – OH / USA<br />

Head and Neck: ................................... Luiz Paulo Kowalski ....................... H. AC Camargo – São Paulo / Brazil<br />

Stomatology: ........................................ Michiel W. M. Van den Brekel ......... Netherlands Cancer Institute – Amsterdam / Netherlands<br />

Pharyngology: ...................................... Marcus Miranda Lessa .................... UFBA – Salvador / Brazil<br />

Laryngology: .......................................... Robert T. Sataloff ............................. Drexel University College of Medicine – Philadelphia / USA<br />

Neurotology: ........................................ Ricardo Ferreira Bento ................... USP– São Paulo / Brazil<br />

Otology: ............................................... Priscila Bogar Rapoport .................. FMABC – Santo André / Brazil<br />

Facial Plastic and Reconstructive: ............. Marcos Mocelin .............................. UFPR – Curitiba / Brazil<br />

Rhinosinusology: .................................. Richard Voegels ............................. USP – São Paulo / Brazil<br />

EDITORIAL BOARD<br />

Adriana Brondani da Rocha – ULBRA - Canoas - Brazil; Adriane Teixeira - UFRGS - Porto Alegre - Brazil; Agrício Crespo - UNICAMP - Campinas - Brazil;<br />

Agustin del Canizo - Universidad de Salamanca - Salamanca - Spain; Alberto Alencar Nuldelmann - PUC - Porto Alegre - Brazil; Alejandro Rivas -<br />

Vanderbilt University Medical Center - Tennessee - USA; Alexandre Felippu Neto - Instituto Felippu - São Paulo - Brazil; Alfio Ferlito - Udine School<br />

of Medicine - Unide - Italy; Ana Cristina H. Hoshino - USP - São Paulo - Brazil; André Luiz Lopes Sampaio - UNB - Brasília - Brazil; Antonio Celso Nassif<br />

Filho - PUC - Curitiba - Brazil; Badr Eldin Mostafa - Ain-Shams University - Cairo - Egypt; Bernard Fraysse - Hôpital PURPAN - Toulouse - France;<br />

Carlos Augusto Pires de Oliveira - UNB - Brasília - Brazil; Carlos Curet - Universidad Nacional de Córdoba - Córdoba - Argentina; Carlos Diógenes<br />

Pinheiro Neto - Albany Medical College - New York - USA; Ciríaco Cristóvão Tavares Atherino - UERJ - Rio de Janeiro -Brazil; Desiderio Passáli -<br />

University Hospital - Siena - Italy; Domenico Cuda - Guglielmo da Saliceto Hospital – Piacenza - Italy; Domingos Hiroshi Tsuji - USP - São Paulo -<br />

Brazil; Eduardo Crema - UFTM – Uberaba - Brazil; Eliane Schochat - USP - São Paulo - Brazil; Elisabeth Carrara de Angelis - Hospital AC Camargo<br />

- São Paulo – Brazil; Fabrizio Ricci Romano - USP - São Paulo - Brazil; Filipe Matuba -Agostinho Neto University - Luanda - Angola; Fernando Luis<br />

Dias - INCA - Rio de Janeiro - Brazil; Francini Grecco de Melo Pádua - UNIFESP - São Paulo - Brazil; Francisco Verissimo de Mello Filho - USP-RP -<br />

Ribeirão Preto - Brazil; Gerson Schulz Maahs - UFRGS - Porto Alegre - Brazil; Giovanni Danesi - Ospedali Riuniti di Bergamo - Bergamo - Italy; Héctor<br />

Rondón Cardoso - Universidad Nacional de San Agustín - Arequipa - Perú; Heinz Stammberger - Graz University - Graz - Austria; Hélio Lessa - UFBA<br />

- Salvador - Brazil; Jacques Magnan - Université dAix-Marseille - Marseille - France; Jair Cortez Mantovani - UNESP - Botucatu - Brazil; Jeferson<br />

S. D’Avila - UFSE - Aracajú - Brazil; Jesús Algaba Guimera - Hospital Donostia de San Sebastián -San Sebastián - Spain; José Faibes Lubianca Neto<br />

- UFCSPA - Porto Alegre - Brazil; Jose N. Fayad - Keck School of Medicine, USC - California - USA; Lídio Granato - FCMSCSP - São Paulo - Brazil; Lilian<br />

Muniz - Universidade Federal de Recife - Recife – Brazil - Luciana Miwa Nita – UNB - Brasília - Brazil; Luiz Antonio Guerra Bernd - UFRS - Porto<br />

Alegre – Brazil; Luiz Lavinsky - UFRGS - Porto Alegre - Brazil; Luiz Ubirajara Sennes - USP - São Paulo - Brazil; Maira Rozenfeld Olchik -UFRGS<br />

- Porto Alegre - Brazil; Manuel Manrique Rodríguez - Universidad de Navarra - Navarra - España; Marcelo Lazzaron Lamers - UFRGS - Porto Alegre<br />

- Brazil; Marcelo Ribeiro de Toledo Piza - Associação Paparella - Ribeirão Preto - Brazil; Márcio Abrahão - UNIFESP - São Paulo - Brazil; Márcio<br />

Nakanishi - UNB - Brasília – Brazil; Marcos Vial Goycoolea - Clinic of Las Condes -Santiago – Chile; Maria Valéria Schimidt Goffi Gómez - USP - São<br />

Paulo – Brazil; Mario Andréa -Lisboa University - Lisboa – Portugal; Mario Svirsky - New York University - New York - USA; Maurizio Barbara -<br />

Sapienza University, SantAndrea Hospital - Rome - Italy; Minoru Hirano -Kurume University - Kurume – Japan; Nédio Steffen - PUC - Porto Alegre<br />

– Brazil; Nelson Rosário - UF do Paraná - Curitiba – Brazil; O. Nuri Özgirgin - Ba_kent University Faculty of Medicine - Ankara – Turkey; Olivier<br />

Sterkers - Université Paris Diderot - Paris – France; Onivaldo Cervantes - UNIFESP - São Paulo – Brazil; Otávio Bejzman Piltcher - UFRGS - Porto<br />

Alegre - Brazil; Paulo Sérgio Lins Perazzo - UNEB - Salvador – Brazil; Pedro L. Coser - UFSM -Santa Maria – Brazil; Pedro Luís Mangabeira Albernaz<br />

- UNIFESP - São Paulo – Brazil; Regina Helena Garcia Martins - UNESP - Botucatu – Brazil; Richard Harvey - University of New South Wales - New<br />

South Wales – Australia; Robert Sweet - McGill University - Montreal – Canada; Robert Vincent - Causse Ear Clinic - Colombiers – France; Roberto<br />

Campos Meirelles - UERJ -Rio de Janeiro – Brazil; Roberto Dihl Angeli - UFRGS - Porto Alegre -Brazil; Roberto Filipo - Sapienza Università di Roma<br />

- Roma – Italy; Rodrigo de Paula Santos - UNIFESP - São Paulo – Brazil; Ronaldo Frizzarini - USP - São Paulo – Brazil; Sady Selaimen da Costa<br />

– UFRGS – Porto Alegre – Brazil; Salvatore Conticello - Università degli Studi di Torino - Turin – Italy; Samir Cahali - HSPE -São Paulo – Brazil;<br />

Shiro Tomita - UFRJ - Rio de Janeiro - Brazil; Silvia Dornelles - UFRGS - Porto Alegre - Brazil; Silvio Antonio Monteiro Marone - PUCCAMP - Campinas<br />

– Brazil; Silvio da Silva Caldas Neto - UFPE - Recife – Brazil; Tania Maria Sih - FMUSP - São Paulo – Brazil; Thomas Linder - Luzerner Kantonsspital<br />

- Luzern – Switzerland; Ugo Fisch - University Hospital - Zürich –Switzerland; Wytske Fokkens - Academic Medical Cente - Amsterdam – Netherlands;<br />

Zelita Ferreira Guedes - UNIFESP - São Paulo - Brazil.<br />

Librarian: Adilson Montefusco<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, Jul/Aug/September - 2013.<br />

235


Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):236.<br />

DOI: 10.7162/S1809-97772013000300001<br />

Editorial<br />

International Archives of Otorhinolaryngology<br />

Editorial<br />

<strong>17</strong> th Volume (3) – Jul/Aug/September - 2013<br />

International Archives of Otorhinolaryngology and Thieme Medical Publishers<br />

On 16 April 2013, the Otorhinolaryngology Foundation signed a partnership with Thieme Medical Publishers for the<br />

publication of the International Archives of Otorhinolaryngology, beginning with this year’s 4th edition.<br />

Thieme, a German publishing company based in Stuttgart with a 125-year history, is one of the most important publishers<br />

of medical books and journals in the world. It publishes 130 diversified medical journals and 70 new books a year.<br />

The International Archives of Otorhinolaryngology is a journal sponsored by the Otorhinolaryngology Foundation and the<br />

Societas Oto-Rhino-Laryngologica Latina, a medical society founded in 1924. It has been continuously published for the last<br />

<strong>17</strong> years and is the first electronic journal of otorhinolaryngology in the world. This scientific journal is reviewed by an<br />

international editorial board and independent peer reviewers and is currently supported by FAPESP (Foundation for<br />

Research Support of the State of Sao Paulo).<br />

This partnership is a major breakthrough for Brazilian science, since our journal, as part of Thieme’s portfolio, will partake<br />

of the publisher’s quality standards and achieve more international visibility.<br />

With this partnership, the International Archives of Otorhinolaryngology will be added to the most important indexations<br />

of medicine in the medium term, thus supplementing its already established academic presence. It will also increase the<br />

contributions from all around the world. This way, we hope the journal will become, soon, the major vehicle for research in the<br />

field of otorhinolaryngology, speech therapy, and related sciences in Latin America. The authors who publish in our journal<br />

will thus have an international impact comparable to researchers whose work appears in other renowned periodicals.<br />

Congratulations to all who have published in our journal and who have induced Thieme, which took notice of the quality<br />

of its contents and its importance among Brazilian experts, to choose it as its starter in Brazil.<br />

Finally, this partnership was made possible by the visionary mission of Professor Ricardo Bento, President of the Board of<br />

Trustees of the Otorhinolaryngology Foundation; the perseverance and encouragement of Professor Richard Voegels; the<br />

institutional and quality commitment of Professor Geraldo Pereira Jotz, Editor-in-Chief; and of Dr. Aline Bittencourt,<br />

Co-Editor; together with the professional dedication of Adilson Montefusco, librarian in charge. This partnership was signed<br />

in Sao Paulo in the presence of Mr. Daniel Schiff, Senior Vice President of Thieme Publishers, and Mrs. Michele Aranha,<br />

representative in Brazil of Thieme Publishers.<br />

For future issues, we will be changing the system of online manuscript submission and review, which will be managed by<br />

ScholarOne (http://mc.manuscriptcentral.com/iaorl)<br />

Best regards,<br />

Geraldo Pereira Jotz<br />

Editor-in-Chief<br />

International Archives of Otorhinolaryngology<br />

Aline Bittencourt<br />

Co-Editor<br />

International Archives of Otorhinolaryngology<br />

Index in LILACS and LILACS-Express – Latindex – DOAJ – FUNPEC-RP –<br />

SciELO – SCOPUS<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, Jul/Aug/September - 2013.<br />

236


ISSN 1809-9777<br />

Contents<br />

Int. Arch. Otorhinolaryngol., <strong>17</strong> (3)<br />

Original Article<br />

242 Tympanomastoidectomy: Comparison between canal wall-down and canal wall-up techniques in<br />

surgery for chronic otitis media<br />

Azevedo AF, Soares ABC, Garchet HQC, Sousa NJA.<br />

246 Endoscopic surgery in the treatment of crista galli pneumatization evolving with localizated frontal<br />

headaches<br />

Socher JA, Santos PG, Correa VC, Silva LCB.<br />

251 Velopharyngeal dysfunction: a systematic review of major instrumental and auditory-perceptual<br />

assessments<br />

Paniagua LM, Signorini AV, Costa SS, Collares MVM, Dornelles S.<br />

257 Results obtained with a low cost software-based audiometer for hearing screening<br />

Ferrari DV, Lopez EA, Lopes AC, Aiello CP, Jokura PR.<br />

265 Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with<br />

cleft lip/palate<br />

Paniagua LM, Signorini AV, Costa SS, Collares MVM, Dornelles S.<br />

274 Clinical and fiberoptic endoscopic assessment of swallowing in patients with chronic obstructive<br />

pulmonary disease<br />

Macri MRB, Marques JM, Santos RS, Furkim AM, Melek I, Rispoli D, Nunes MCA.<br />

279 Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx<br />

Ido Filho JM, Carvalho B, Mizoguchi FM, Catani GSA, Macedo Filho ED, Malafaia O,Stahlke Jr. HJ.<br />

285 Audiological outcomes of cochlear implantation in Waardenburg Syndrome<br />

Magalhães ATM, Samuel PA, Gomez MVSG, Tsuji RK, Brito B, Bento RF.<br />

291 Performance analysis of ten brands of batteries for hearing aids<br />

Penteado SP, Bento RF.<br />

305 Contribution of audiovestibular tests to the topographic diagnosis of sudden deafness<br />

Oiticica J, Bittar RSM, Castro CC, Grasel S, Pereira LV, Bastos SL, Ramos ACM, Beck R.<br />

315 Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy for botulinum toxin injection:<br />

an experimental study in cadaver larynges<br />

Alonso VMO, Chagury AA, Hachiya A, Imamura R, Tsuji DH, Sennes LU.<br />

321 Correlation of cephalometric and anthropometric measures with obstructive sleep apnea severity<br />

B<strong>org</strong>es PTM, Ferreira Filho ES, Araujo TME, Moita Neto JM, B<strong>org</strong>es NES, Melo Neto B, Campelo V, Paschoal JR, Li LM.<br />

Review Article<br />

329 Use of surface electromyography in phonation studies: an integrative review<br />

Balata PMM, Silva HJ, Moraes KJR, Pernambuco LA, Moraes SRA.<br />

340 Middle ear adenoma with neuroendocrine differentiation: relate of two cases and literature review<br />

Bittencourt AG, Tsuji RK, Cabral Junior F, Pereira LV, Fonseca ACO, Alves V, Bento RF.<br />

Case Report<br />

344 Bullous Systemic Lupus Erythematosus: Case report<br />

Miziara ID, Mahmoud A, Chagury AA, Alves RD.<br />

347 Eagle’s Syndrome<br />

Pinheiro TG, Soares VYR, Ferreira DBL, Raymundo IT, Nascimento LA, Oliveira CACP.<br />

351 Retrolabyrinthine approach for cochlear nerve preservation in neurofibromatosis type 2 and simultaneous<br />

cochlear implantation<br />

Bento RF, Monteiro TA, Bittencourt AG, Gomez MVSG, Brito R.<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, Jul/Aug/September - 2013.<br />

237


AUTHOR GUIDELINES<br />

INTERNATIONAL ARCHIVES OF<br />

OTORHINOLARYNGOLOGY<br />

Editor-in-Chief - Geraldo Pereira Jotz, M.D. Ph.D.<br />

Co- Editor - Aline Bittencourt, M.D.<br />

Editorial Office:<br />

Rua Teodoro Sampaio 483<br />

Zip code 05405-000<br />

São Paulo – SP – Brazil<br />

Phone/FAX: +55 (11) 3085-9943<br />

archives@internationalarchivesent.<strong>org</strong><br />

International Archives of Otorhinolaryngology (IAO)<br />

is an international peer-reviewed journal dedicated to the<br />

otolaryngology–head and neck surgery, audiology and speech<br />

therapy.<br />

IAO is published every three months and supports the<br />

World Health Organization (WHO) and of the International<br />

Committee of Medical Journal Editors (ICMJE) politics regarding<br />

registration of clinical trials. Therefore from now on we will<br />

only accept for publication articles of clinical trials that have<br />

been given a number of identification from one of the Clinical<br />

Essay Registry validated by the criteria established by the<br />

WHO and the ICMJE, which links are available at the ICMJE<br />

(http://www.icmje.<strong>org</strong>/). The identification number should<br />

be informed at the end of the abstract.<br />

IAO reserves the right to exclusive publication of all<br />

accepted manuscripts. We will not consider any manuscript<br />

previously published nor under review by another publication.<br />

Once accepted for review, the manuscript must not be<br />

submitted elsewhere. Transfer of copyright to IAO is a<br />

prerequisite of publication. All authors must sign a copyright<br />

transfer form.<br />

Authors must disclose any financial relationship(s) at<br />

the time of submission, and any disclosures must be updated<br />

by the authors prior to publication. Information that could be<br />

perceived as potential conflict(s) of interest must be stated.<br />

This information includes, but is not limited to, grants or<br />

funding, employment, affiliations, patents, inventions,<br />

honoraria, consultancies, royalties, stock options/ownership,<br />

or expert testimony.<br />

Article Categories<br />

The journal publishes the types of articles defined<br />

below. When submitting your manuscript, please follow the<br />

instructions relevant to the applicable article category.<br />

Original Research: Original, in-depth, clinical or<br />

basic science investigations that aim to change clinical<br />

practice or the understanding of a disease process. Article<br />

types include, but are not limited to, clinical trials, before-andafter<br />

studies, cohort studies, case-control studies, crosssectional<br />

surveys, and diagnostic test assessments. Components<br />

of original research are:<br />

• A title page, including the manuscript title and all authors’<br />

full names, academic degrees (no more than three),<br />

institutional affiliations, and locations. Designate one<br />

author as the corresponding author. Also indicate where<br />

the paper was presented, if applicable.<br />

• A structured Abstract of up to 250 words with the headings:<br />

Introduction, Objective, Methods, Results, and Conclusion.<br />

• The Manuscript body should be divided as: introduction<br />

with objective(s); method; result; discussion; conclusion;<br />

references.<br />

• Manuscript length of no more than 24 pages (exclusive of<br />

the title page and abstract). There is no limit on references.<br />

• Studies involving human beings and animals should<br />

include the approval protocol number of the respective<br />

Ethics Committee on Research of the institution from<br />

which the research is affiliated.<br />

Systematic Reviews (including Meta-analyses):<br />

Critical assessments of literature and data sources on<br />

important clinical topics in otolaryngology-head and neck<br />

surgery. Systematic reviews that reduce bias with explicit<br />

procedures to select, appraise, and analyze studies are<br />

highly preferred over traditional narrative reviews. The<br />

review may include a meta-analysis, or statistical synthesis<br />

of data from separate, but similar, studies leading to a<br />

quantitative summary of the pooled results. The components<br />

of a systematic review are:<br />

• A title page, including the manuscript title and all authors’<br />

full names, academic degrees, institutional affiliations,<br />

and locations. Designate one author as the corresponding<br />

author. Also indicate where the paper was presented, if<br />

applicable.<br />

• A structured Abstract of up to 250 words with the headings:<br />

Introduction, Objectives, Data Synthesis and Conclusion.<br />

• The Manuscript body should be divided as: introduction;<br />

review of literature; discussion; final comments; references.<br />

• Manuscript length of no more than 24 pages (exclusive of<br />

the title page and abstract). There is no limit on references.<br />

Case Reports: Report of a truly unique, highly relevant,<br />

and educationally valuable case.<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, Jul/Aug/September - 2013.<br />

238


• A title page, including the manuscript title and all authors’<br />

full names, academic degrees, institutional affiliations,<br />

and locations. Designate one author as the corresponding<br />

author. Also indicate where the paper was presented, if<br />

applicable.<br />

• A structured Abstract of up to 250 words with the headings:<br />

Introduction, Objectives, Resumed Report and Conclusion.<br />

• The Manuscript body should be divided as: introduction;<br />

review of literature with differential diagnosis; case report;<br />

discussion; final comments; references.<br />

• Manuscript length: no more 2 pages.<br />

• The Manuscript should include the approval protocol<br />

number of the respective Ethics Committee on Research of<br />

the institution from which the research is affiliated.<br />

In accordance with double-blind review, author/<br />

institutional information should be omitted or blinded from<br />

the following submission files: Manuscript, Figure(s), Table(s),<br />

Response to Reviewers.<br />

The Abstract should be followed by three to six<br />

keywords in English, selected from the list of Descriptors<br />

(Mesh) created by National Library of Medicine and available<br />

on http://www.nlm.nih.gov/mesh/2013/mesh_browser/<br />

MBrowser.html.<br />

Abbreviations: Do not use abbreviations in the title or<br />

abstract. When using abbreviations in the text, indicate the<br />

abbreviation parenthetically after the first occurrence and use<br />

the abbreviation alone for all subsequent occurrences.<br />

Update Manuscripts: The manuscript is an update<br />

that explores a particular subject, developed from current<br />

data, based on recently published works.<br />

• A title page, including the manuscript title and all authors’<br />

full names, academic degrees, institutional affiliations,<br />

and locations. Designate one author as the corresponding<br />

author. Also indicate where the paper was presented, if<br />

applicable.<br />

• A structured Abstract of up to 250 words with the headings:<br />

Introduction, Objectives, Data Synthesis and Conclusion.<br />

• The Manuscript body should be divided as: introduction;<br />

review of a particular subject; discussion; final comments;<br />

references.<br />

• Manuscript length of no more than 15 pages (exclusive of<br />

the title page and abstract). There is no limit on references.<br />

Letters to the Editor and Opinion articles: Only by<br />

invitation from the Editorial Board. Manuscript length: no<br />

more 2 pages.<br />

Manuscript Preparation<br />

Correct preparation of the manuscript will expedite the<br />

review and publishing process. Manuscripts must conform to<br />

acceptable English usage.<br />

Necessary Files for Submission (each topic should start<br />

in a new page):<br />

• Title Page<br />

• Abstract<br />

• Manuscript (main text, references and figure legends)<br />

• Figure(s) (when appropriate)<br />

• Table(s) (when appropriate)<br />

Authorship: Authorship credit should be based on<br />

criteria established by the International Committee of Medical<br />

Journal Editors: 1) substantial contributions to conception<br />

and design, acquisition of data, or analysis and interpretation<br />

of data; 2) drafting the article or revising it critically for<br />

important intellectual content; and 3) final approval of the<br />

version to be published.<br />

References: Authors are responsible for the<br />

completeness, accuracy, and format of their references.<br />

References should be numbered consecutively as they are<br />

cited in Arabic numbers the text between parentheses. All<br />

authors shall be listed in full up to the total number of six; for<br />

seven or more authors, list the first six authors and add “et al.”.<br />

There should be no more than 90 references for Original<br />

Articles, 120 for Literature review or update articles and 15 for<br />

Case Report articles. Refer to the List of Journals Indexed in<br />

Index Medicus for abbreviations of journal names, or access<br />

the list at http://www.nlm.nih.gov/tsd/serials/lji.html.<br />

Sample references are given below. For more information,<br />

please check: http://www.ncbi.nlm.nih.gov/books/NBK7256.<br />

Examples:<br />

- Journals: Author | Article Title | Journal Title |<br />

Date of Publication | Volume Number | Issue Number |<br />

Pagination.<br />

Huttenhower C, Gevers D, Knight R, et al. Structure,<br />

function and diversity of the healthy human microbiome.<br />

Nature. 2012;486(7402):207-14.<br />

- Dissertations and Theses: Author | Title | Content<br />

Type | Place of Publication | Publisher | Date of Publication<br />

| Pagination.<br />

Baldwin KB. An exploratory method of data retrieval<br />

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Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, Jul/Aug/September - 2013.<br />

241


Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):242-245.<br />

DOI: 10.7162/S1809-97772013000300002<br />

Tympanomastoidectomy: Comparison between canal wall-down and canal<br />

wall-up techniques in surgery for chronic otitis media<br />

Alexandre Fernandes de Azevedo 1 , Anna Bárbara de Castro Soares 2 , Henrique Queiroz Correa Garchet 3 ,<br />

Nicodemos José Alves de Sousa 4 .<br />

1) MD in Infectology at the Federal University of Minas Gerais. Assistant Doctor - Otolaryngology Clinic of Santa Casa B.H.<br />

2) Resident of Plastic Surgery at the Hospital Mater Dei.<br />

3) Resident of Otolaryingology at the Santa Casa B.H.<br />

4) M.D. in Otolaryngology at the Federal University of São Paulo. Honorary Chief of the Otolaryngology Clinic of Santa Casa B.H.<br />

Institution: Santa Casa of Belo Horizonte<br />

Belo Horizonte / MG - Brazil.<br />

Mailing address: Clínica de Otorrinolaringologia da Santa Casa de Belo Horizonte - Alexandre Fernandes de Azevedo - Rua Perdigão Malheiro, 195/ 501 - Cidade Jardim<br />

- Belo Horizonte / MG - Brazil - Zip code: 30380-050 - Telephone: (+55 31) 8863-8548 - E-mail: xandefa@hotmail.com<br />

Article received on September 2 nd , 2012. Article accepted on April 15 th , 2013.<br />

SUMMARY<br />

Introduction: Chronic otitis media (COM) is an inflammatory condition associated with otorrhea as well as large and persistent<br />

perforations of the tympanic membrane in some cases. COM can also lead to cholesteatoma. Surgical treatment with canal walldown<br />

and canal wall-up tympanomastoidectomy is considered for both types of illness. The choice of technique is controversial<br />

and is dependent on several factors, including the extent of disease.<br />

Objective: We aimed to evaluate surgical outcomes in COM patients with and without cholesteatoma treated with canal walldown<br />

and canal wall-up tympanomastoidectomy. Disease eradication and post-operative auditory thresholds were assessed.<br />

Method: Patient records from the otorhinolaryngology department of a tertiary hospital were assessed retrospectively.<br />

Results: Patients who underwent canal wall-up tympanomastoidectomy had a higher rate of revision surgery, especially those<br />

with cholesteatoma. However, there were no statistically significant differences in post-operative hearing thresholds between<br />

the two techniques.<br />

Conclusion: The canal wall-down technique is superior to the canal wall-up technique, especially for patients with cholesteatoma.<br />

Keywords: Chronic Disease; Otitis Media; Hearing Loss; Cholesteatoma, Middle Ear; Reoperation.<br />

INTRODUCTION<br />

Otitis media is defined as an inflammatory disease<br />

of the middle ear that may be infectious or not and focal<br />

or generalized. The course of disease may be acute with<br />

a tendency towards total resolution and a return to the<br />

integrity of the regions affected, or it may be chronic with<br />

permanent sequelae (1,2,3).<br />

Chronic otitis media (COM) is clinically characterized<br />

as an inflammatory condition associated with otorrhea and<br />

tympanic membrane perforation in some cases. The<br />

disease course is more than 3 months in duration and<br />

histopathologically it is associated with irreversible tissue<br />

changes.<br />

The incidence of COM is higher in less developed<br />

countries. Malnutrition, poor hygiene, poor quality<br />

housing, and high population density are factors that are<br />

associated with a higher incidence of middle ear infections<br />

(3,4).<br />

COM can be subdivided into two groups:<br />

cholesteatomatous chronic otitis media (CCOM) and<br />

chronic otitis media without cholesteatoma (COMWC). A<br />

central or marginal perforation may be present. The<br />

inflammatory process in the middle ear mucosa may show<br />

different stages of evolution.<br />

CCOM is characterized by epithelial accumulation<br />

with keratin production in the middle ear. Cholesteatoma<br />

may be classified as congenital or acquired, and is further<br />

categorized as primary or secondary cholesteatoma. Clinical<br />

and surgical treatments are available for COM. The first is<br />

reserved for COMWC when patient follow-up is possible.<br />

The surgical approach is suitable for both CCOM and<br />

COMWC and encompasses tympanoplasty, canal wall-up<br />

(CWU) and canal wall-down (CWD) mastoidectomy (1,5,6)<br />

and its variations, including modified radical mastoidectomy<br />

or Bondy’s procedure. The choice of technique remains<br />

controversial and is usually decided based on the presence<br />

or absence of cholesteatoma, its location, the state of the<br />

middle ear mucosa, and auditory thresholds. Recurrence and<br />

post-operative functional status vary between techniques.<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 242-245, Jul/Aug/September - 2013.<br />

242


Tympanomastoidectomy: Comparison between canal wall-down and canal wall-up techniques in surgery for chronic otitis media.<br />

Azevedo et al.<br />

The aim of this study was to clarify which surgical<br />

technique provides the best outcomes in terms of disease<br />

control and improved hearing thresholds.<br />

METHOD<br />

This was a retrospective study of an historical<br />

cohort. The medical records of patients with COM who<br />

underwent a CWU or CWD mastoidectomy at the<br />

otorhinolaryngology department of a tertiary hospital<br />

between 1997 and 2005 were evaluated.<br />

Postoperative outcomes for the 2 techniques<br />

mentioned above were compared using control of the<br />

disease, absence of otorrhea, and cholesteatoma recurrence<br />

during the follow-up period, which was at least 24 months,<br />

as criteria. Pure tone average hearing thresholds at 500 Hz,<br />

1000 Hz, and 2000 Hz were also compared before and<br />

after surgery for both techniques.<br />

Statistical analyses were performed using the Chi<br />

square test, and p values


Tympanomastoidectomy: Comparison between canal wall-down and canal wall-up techniques in surgery for chronic otitis media.<br />

Azevedo et al.<br />

DISCUSSION<br />

Among the patients with COMWC, the disease<br />

control rate was 91.9%, regardless of the technique used,<br />

which is similar to the rates reported in other studies, which<br />

have ranged from 63% to 96% (7,8,9,10). Among the<br />

patients with CCOM, the disease control rate was 64.1%,<br />

which is slightly lower than previous reports, which have<br />

ranged from 75% to 90% (7,11,12,13).<br />

When the CWU technique was used, the disease<br />

control rate for the first surgery was 76.6%. In contrast,<br />

when the CWD technique was used, the disease control<br />

rate was 85.7%, regardless of the presence of cholesteatoma.<br />

Data in the literature are similar with reported values<br />

ranging from 71% to 95% for the CWU technique<br />

(9,11,14,15,16) and from 71% to 96% for the CWD<br />

technique (9,11,13,14,15,16).<br />

In the COMWC group, a higher rate of revision<br />

surgery was found among patients who underwent a CWD<br />

mastoidectomy (25%) compared with a CWU<br />

mastoidectomy (12.2%). This can be explained by the fact<br />

that patients with more severe disease were selected for<br />

CWD mastoidectomy.<br />

Of the patients with CCOM who underwent a CWU<br />

mastoidectomy, 57.9% required revision surgery whereas<br />

only 15% of those who underwent a CWD mastoidectomy<br />

required revision surgery. The current literature also shows<br />

higher recurrence rates when patients with cholesteatoma<br />

undergo a CWU mastoidectomy. Cruz et al. (2001) reported<br />

surgical revision rates of 37.5% and 26.08% when using the<br />

CWU and CWD techniques, respectively. We believe that<br />

in our study the higher rate of reoperation observed when<br />

preserving the canal wall is related to the longer follow-up<br />

(median 7.5 years), and suggests late complications of the<br />

disease, which are not uncommon when the CWU technique<br />

is used.<br />

The choice of technique remains controversial but<br />

this study, in agreement with the literature, has shown that<br />

cholesteatoma can be treated with the CWU technique.<br />

However, Bento et al. and Cruz et al. (14,5) suggest that<br />

criteria such as cholesteatoma restricted to the attic, good<br />

condition of the middle ear mucosa, and the possibility of<br />

good postoperative follow-up are required before the<br />

CWU technique is used.<br />

In this study, no statistically significant difference in<br />

pure tone average thresholds before and after surgery with<br />

either of the techniques. Because patients with less than 2<br />

years follow-up were excluded from the study, there was<br />

a considerable decrease in the number of individuals<br />

available for analysis, which hindered any robust analysis of<br />

this variable. In the literature we found many studies that<br />

reported better audiometric results when the CWU technique<br />

was used rather than the CWD technique (7,8,10,11,15,<strong>17</strong>).<br />

However, other studies have reported no significant<br />

differences in hearing outcomes in association with the two<br />

techniques (16).<br />

CONCLUSION<br />

The CWD technique and its various modifications<br />

results in better outcomes, especially when it comes to<br />

surgery to control CCOM. Some precautions facilitate a<br />

satisfactory functional outcome with better control of<br />

persistent otorrhea and greater certainty as to the<br />

eradication of cholesteatoma compared to CWU<br />

mastoidectomy.<br />

REFERENCES<br />

1. Costa SS, Dornelles CC, Netto LFS, Braga MEL. Aspectos<br />

gerais das otites médias. In: Costa SS, Cruz OLM, Oliveira<br />

JAA. Otorrinolaringologia Princípios e Prática. 2nd ed. São<br />

Paulo: Artmed; 2006. p. 254-73.<br />

2. Bluestone CD. Epidemiology and pathogenesis of<br />

chronicsuppurative otitismedia: implications for prevention<br />

and treatment. Int J Pediatr Otorhinolaryngol. 1998<br />

Jan;420:207-23.<br />

3. Azevedo AF, Pinto DCG, Souza NJA, Greco DB, Gonçalves<br />

DU. Perda auditiva sensório-neural na otite média crônica<br />

supurativa em pacientes com e sem colesteatoma. Braz J<br />

Otorhinolaryngol. 2007 Sep-Oct;73(5):671-4.<br />

4. Godinho RN, Goncalves TM, Nunes FB, et al. Prevalence<br />

and impact of chronicotitismedia in school age children in<br />

Brazil. First epidemiologic study concerning<br />

chronicotitismedia in Latin America. Int J Pediatr<br />

Otorhinolaryngol. 2001 Dec;61(3):223-32.<br />

5. Cruz OLM, Campos CAH. Cirurgia para a otite média<br />

crônica. Acta Otorrinolaringol. 2005;23(1):33-8.<br />

6. Roland PS, Isaacson B, Kutz JW. Office management of<br />

tympanic membrane perforation and the draining ear. In:<br />

Brackmann DE, Shelton C, Arriaga MA. Otologic Surgery.<br />

3rd ed. Philadelphia: Saunders Elsevier; 2010. p. 107-18.<br />

7. Segalla DK, Nakao LH, Anjos MF, Penido NO. Resultados<br />

cirúrgicos e audiológicos pós mastoidectomia em um serviço<br />

de Residência Médica. Acta Otorrinolaringol.<br />

2008;26(3):<strong>17</strong>8-81.<br />

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Tympanomastoidectomy: Comparison between canal wall-down and canal wall-up techniques in surgery for chronic otitis media.<br />

Azevedo et al.<br />

8. Vartianinen E, Nuutinen J. Long term hearing results of<br />

one stage tympanoplasty for chronic otitis media. Eur Arch<br />

Otorhinolaryngol. 1992;249(6):329-31.<br />

9. Veldman JE, Braunius WW. Revision surgery for chronic<br />

otitis media: a learning experience. Report on 389 cases<br />

with a long-term follow-up. Ann Otol Rhinol Laryngol. 1998<br />

Jun;107(6):486-91.<br />

10. Cruz OL, Kasse CA, Leonhart FD. Efficacy of surgical<br />

treatment of chronic otitis media. Otolaryngol Head Neck<br />

Surg. 2003 Feb;128(2):263-6.<br />

11. Cruz OLM, Kasse CA, Leonhardt FD. Eficácia do tratamento<br />

cirurgico da otite média crônica colesteatomatosa. Braz J<br />

Otorhinolaryngol. 2001 Mar-Apr;67(2):142-6.<br />

12. Cook JA, Krishnan S, Fagan PA. Hearing results following<br />

modified radical versus canalup mastoidectomy. Ann Otol<br />

Rhinol Laryngol. 1996 May;105(5):379-83.<br />

13. Chang CC, Chen MK. Canal-wall-down tympanoplasty<br />

with mastoidectomy for advanced cholesteatoma. J<br />

Otolaryngol. 2000 Oct;29(5):270-3.<br />

14. Bento RF, Rezende VA, Soares IP. Mastoidectomia: Estado<br />

atual da indicação cirúrgica do ponto de vista infeccioso.<br />

Braz J of Otorhinolaryngol. 2004 Apr-Jun;60(2):98-102.<br />

15. Harkness P, Brown P, Fowler S, Grant H, Ryan R, Topham<br />

J. Mastoidectomy audit: results of the Royal College of<br />

Surgeons of England comparative audit of ENT surgery. Clin<br />

Otolaryngol Allied Sci. 1995 Feb;20(1):89-94.<br />

16.Zhang X, Chen Y, Liu Q, Han Z, Xu A, Ding Y. Long-term<br />

results analysis of mastoidectomy for chronic otitis media. Lin<br />

Chuang Er Bi Yan Hou Ke Za Zhi. 2005 Oct;19(19):870-2.<br />

<strong>17</strong>. Murphy TP, Wallis DL. Hearing results in pediatric patients<br />

after canal-wall-up and canal-wall-down mastoid surgery.<br />

Otolaryngol Head Neck Surg. 1998 Nov;119(5):439-43.<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 242-245, Jul/Aug/September - 2013.<br />

245


Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):246-250.<br />

DOI: 10.7162/S1809-97772013000300003<br />

Endoscopic surgery in the treatment of crista galli pneumatization evolving<br />

with localizated frontal headaches<br />

Jan Alessandro Socher 1 , Pedro Geisel Santos 2 , Vinicius Cidral Correa 3 , Leandro Caetano de Barros e Silva 3 .<br />

1) Doctor of Otolaryngology Faculty of Medicine, University of Sao Paulo. Professor of Otorhinolaryngology in FURB - Regional University of Blumenau.<br />

2) Otolaryngologist.<br />

3) Student of Medicine in FURB - Regional University of Blumenau.<br />

Institution: FURB - Regional University of Blumenau.<br />

Blumenau / SC – Brazil.<br />

Mailing address: Jan Alessandro Socher - Alameda Duque de Caxias, 145 - sala 306 - Bairro Centro – Zip code: 89015-010 - Blumenau / SC – Brazil - E-mail:<br />

jan_socher@yahoo.com.br<br />

Article received on November 19 th , 2012. Article accepted on March 19 th , 2013.<br />

SUMMARY<br />

Introduction: The crista galli is part of the ethmoid bone and thus may suffer from the process of pneumatization. Pneumatization<br />

occurs in between 3% and 14% of patients, resulting from air cells in the frontal or ethmoid sinuses.<br />

Aim: To describe 3 cases of crista galli pneumatization in which the patients developed infection and were treated surgically<br />

by endoscopic techniques.<br />

Method: We present 3 case studies of patients complaining of severe frontal headaches. The patients underwent ENT evaluation,<br />

examination by video-endoscopy, and computed tomography, which identified crista galli pneumatization with mucosal thickening<br />

and the presence of fluid. Patients underwent treatment with antibiotics and corticosteroids; however, they showed no symptomatic<br />

improvement, displayed recurrence of symptoms, and maintained radiographic changes. Thus, patients then underwent drainage<br />

through the crista galli via an endoscopic procedure.<br />

Discussion: During surgery, mucopurulence and/or mucosal thickening and edema were identified in the pneumatized crista<br />

galli. There were no complications during or after surgery. Postoperatively, headache was improved in patients after a minimum<br />

follow-up of 6 months.<br />

Conclusion: Crista galli pneumatization can result in infection, simulating rhinosinusitis. When there is little response to drug<br />

therapy, endoscopic surgical treatment is required; the current cases demonstrate that this technique is safe and effective.<br />

Keywords: Natural Orifice Endoscopic Surgery; Video-Assisted Surgery; Headache.<br />

INTRODUCTION<br />

The crista galli lies on the midline of the cribriform<br />

plate. The falx cerebri attaches anteriorly tothis bone<br />

formation a thin posterior border and slightly curve , and<br />

the anterior border is attached to the frontal bone completing<br />

the margin of the foramen cecum. The crista galli is<br />

embryologically derived from the ethmoid bone (1).<br />

Regarding pneumatization of the crista galli, 2 theories are<br />

valid: pneumatization can originate from the ethmoid sinus<br />

or the frontal sinus. The oldest theory in which the<br />

pneumatization result from the ethmoid sinus is based on<br />

the embryological origin of the crista galli, the ethmoid<br />

bone, and states that the displacement of ethmoidal air<br />

cells would lead to increased aeration of the crista galli 2 . In<br />

the theory explaining pneumatization by the frontal sinus,<br />

sinus extension is likely to cause increased aeration beyond<br />

the normal margin of the frontal bone (3). The incidence<br />

of crista galli pneumatization has been reported to be<br />

between 2.8% and 14.1%, depending on the population<br />

studied (2,4,5). The possibility of involvement of<br />

inflammatory and/or infectious processes in pneumatization<br />

of the crista galli is a noteworthy finding; however, such<br />

involvement is very rare since there are no reports of it in<br />

the literature.<br />

AIM<br />

The aim of this study was to describe 3 cases where<br />

crista galli pneumatization evolved into inflammation and<br />

infection and was treated surgically by endoscopic<br />

techniques.<br />

CASE STUDY #1<br />

A 57-year-old female patient sought advice from<br />

the Specialized Service in Otolaryngology in June 2008,<br />

complaining of a frontal headache that had been present<br />

for the last year. The patient reported previous drug<br />

treatments for sinusitis including azithromycin for 5 days,<br />

amoxicillin for 10 days, and a combination of amoxicillin<br />

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Figure 1. Preoperative coronal section CT scan showing crista<br />

galli pneumatization with mucosal thickening.<br />

Figure 2. Video-endoscopic visualization of the left nasal<br />

cavity showing the septal incision site and opening of the<br />

bone wall of the pneumatized crista galli. SEPTO = nasal<br />

septum; CM = middle turbinate; CI = inferior turbinate.<br />

and clavulanate potassium for 10 days. However, she<br />

always experienced recurrence and progressive worsening<br />

of her symptoms. Examination with video-endoscopy<br />

showed hypertrophy of the lower and middle turbinates<br />

bilaterally associated with septal deviation in the left nasal<br />

cavity. A computed tomography (CT) scan of the paranasal<br />

sinuses and nasal cavity in the axial, coronal, and sagittal<br />

planes with a bone window of 2500–3500 rads was<br />

requested, which identified pneumatization of the crista<br />

galli with mild mucosal thickening in the interior (Figure 1).<br />

This finding indicated endoscopic surgery through the<br />

transseptal approach in the left nasal cavity for drainage<br />

and cleaning of the pneumatized crista galli (Figure 2).<br />

CASE STUDY #2<br />

A 35-year-old female patient sought advice from<br />

the Specialized Service in Otolaryngology in August 2010,<br />

complaining of a localized headache, cacosmia, and nasal<br />

obstruction with progressive worsening of her symptoms<br />

during the previous 6 months. Her symptoms had remained<br />

after drug treatment with amoxicillin and clavulanate<br />

potassium for 10 days. Video-endoscopy showed a deviated<br />

septum and inferior turbinate hypertrophy. A CT scan of<br />

the paranasal sinuses and nasal cavity in the axial, coronal,<br />

and sagittal planes with a bone window of 2500–3500 rads<br />

was requested, which identified pneumatization of the<br />

crista galli apophysis with signs of mucosal thickening and<br />

obliteration interiorly. We introduced antibiotic treatment<br />

(levofloxacin at a dose of 500 mg/day) for 14 days,<br />

corticosteroids (prednisone at a dose of 40 mg/day) for 7<br />

days, and symptomatic analgesia. The patient’s symptoms<br />

Figure 3. Coronal CT section showing mucosal thickening<br />

and obliteration of the pneumatized crista galli.<br />

improved temporarily, but 5 days after treatment with<br />

antibiotics was stopped, recurrence of her symptoms<br />

occurred. A second CT scan of the paranasal sinuses and<br />

nasal cavity showed the maintenance of mucosal thickening<br />

and obliteration within the crista galli (Figure 3). Endoscopic<br />

surgery by a transseptal approach through the right nostril<br />

was indicated for drainage and cleaning of the pneumatized<br />

crista galli (Figures 4-7).<br />

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Socher et al.<br />

Figure 4. Intraoperative endoscopic visualization of the right<br />

nasal cavity showing septal incision and detachment.<br />

SEPTO = nasal septum; CM = middle turbinate.<br />

Figure 5. Intraoperative endoscopic visualization of the right<br />

nasal cavity demonstrating septal resection to allow approach<br />

to the bone wall of the pneumatized crista galli.<br />

SEPTO = nasal septum; CM = middle turbinate.<br />

Figure 6. Intraoperative endoscopic visualization of the right<br />

nasal cavity showing transseptal opening of the bone wall of<br />

the pneumatized crista galli.<br />

SEPTO = nasal septum; CM = middle turbinate.<br />

Figure 7. Intraoperative endoscopic visualization of the right<br />

nasal cavity demonstrating the detail of opening of the<br />

pneumatized crista galli, the lining of which shows mucosal<br />

thickening and edema.<br />

CASE STUDY #3<br />

A 31-year-old male patient sought advice from the<br />

Specialized Service in Otolaryngology in April 2012, with<br />

the complaint of chronic nasal obstruction that had been<br />

present since childhood and was associated with the<br />

symptoms of congestion, rhinorrhea, and facial pain<br />

localized in the frontal region for the last 3 months. He had<br />

shown no improvement after 2 previous episodes of<br />

treatment with antibiotics (levofloxin and amoxicillin<br />

lasting 10 days and 14 days, respectively). He also<br />

reported a history of adenoidectomy surgery prior to the<br />

age of 5. Video-endoscopic examination identified the<br />

presence of septal deviation in the left nasal cavity<br />

associated with mild inferior and right middle turbinate<br />

hypertrophy as well as hypertrophy of lymphoid tissue in<br />

the nasopharynx. A sinus and nasal cavity CT scan in the<br />

axial, coronal, and sagittal planes was requested, which<br />

identified septal deviation with bone spur formation,<br />

prominence of the soft parts of the cavum, and<br />

pneumatization of the crista galli with significant mucosal<br />

thickening and fluid present within (Figure 8). The<br />

patient underwent surgical resection of the lymphoid<br />

tissue present in the nasopharynx and transseptal<br />

endoscopic drainage of the pneumatized crista galli.<br />

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Socher et al.<br />

another 6 months thereafter. These follow-ups ensured<br />

maintenance of the patency of the crista galli through<br />

video-endoscopic examinations. After 6 months, the patients<br />

were advised to return annually or if there was a need.<br />

The collected material was sent for bacterial and<br />

fungal culture, but the results were inconclusive, which can<br />

be explained by use of antibiotics prior to surgery. The use<br />

of different antibiotics prescribed based on clinical criteria<br />

for sinusitis was common in the history of all the patients<br />

prior to surgery. However, the use of antimicrobial agents<br />

was apparently unsuccessful in treating this condition, in<br />

view of the maintenance or recurrence of symptoms in all<br />

patients.<br />

Figure 8. Coronal CT section showing mucosal thickening<br />

and fluid within the pneumatized crista galli.<br />

DISCUSSION<br />

Most studies on the crista galli in the literature are<br />

anatomical descriptions from surveys of series of CT scans<br />

or findings obtained during access to the anterior skull base<br />

using endoscopic surgery (1-5). There is a predominance<br />

in the study of pneumatization of the crista galli in females,<br />

with a ratio of 2 females:1 male between the ages of 31 and<br />

57 years. However, reports on pneumatization of the crista<br />

galli related to the complaint of headache were not found<br />

in the literature, and neither was evidence of the possibility<br />

of infection from such pneumatization.<br />

In the present study, it was possible to access<br />

pneumatization of the crista galli and confirm the findings<br />

of CT scans showing signs of inflammation and/or infection<br />

in all 3 cases by using a transseptal endoscopic approach.<br />

In the first patient, mucosal thickening and edema was<br />

identified, while the second and third cases were associated<br />

with purulent drainage and identification of mucosal<br />

thickening and edema within the pneumatized crista galli.<br />

There were no transoperative or postoperative<br />

complications. However, it is important to note that good<br />

knowledge of the field of endoscopic technique and the<br />

anatomy of this region is essential, especially areas near the<br />

crista galli such as the cribriform plate and other structures<br />

of the anterior skull base. Patients were discharged on the<br />

same day as surgery, about 8 hours after operation.<br />

Antibiotics (oral levofloxacin at a dose of 500 mg/day for<br />

10 days in the first case and oral clindamycin at a dose of<br />

900 mg/day for 10 days in the other cases) plus<br />

corticosteroids (oral prednisone at a dose of 40 mg/day for<br />

7 days) were administered. Postoperative follow-ups were<br />

performed weekly during the first month and monthly for<br />

The findings of video-endoscopy examinations were<br />

nonspecific and did not contribute significantly to the<br />

diagnosis of these cases. The suspected inflammation and/<br />

or infection within the pneumatized crista galli were only<br />

confirmed by CT scan.<br />

In all cases, pathological results confirmed the<br />

presence of fragments of upper airway tract mucosa that<br />

were lined by pseudostratified ciliated epithelium resting<br />

on corium and contained a discrete amount of mononuclear<br />

inflammatory cells and eosinophils, which is characteristic<br />

of chronic inflammation of the respiratory mucosa contained<br />

inside the crista galli. Furthermore, pathological examination<br />

in the third patient confirmed the presence of lymphoid<br />

tissue in the nasopharynx that had no signs of malignancy<br />

and was compatible with pharyngeal tonsil.<br />

The patients complained of transient postoperative<br />

nasal obstruction and hyposmia/anosmia, but showed<br />

improvement of symptoms and had no recurrence during<br />

a clinical monitoring period of 3 years in the first patient,<br />

1 year and 6 months in the second patient, and 6 months<br />

in the third patient.<br />

CONCLUSION<br />

Pneumatization of the crista galli may be complicated<br />

by inflammatory and/or infectious processes simulating<br />

rhinosinusitis and shows little response to drug therapy,<br />

thus requiring endoscopic surgical treatment. In the current<br />

cases, such treatment was demonstrated to be safe and<br />

effective.<br />

REFERENCES<br />

1. Som PM, Park EE, Naidich TP, Lawson W. Crista Galli<br />

Pneumatization Is an Extension of the Adjacent Frontal<br />

Sinuses. AJNR Am J Neuroradiol. 2009;30:31–3.<br />

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Endoscopic surgery in the treatment of crista galli pneumatization evolving with localizated frontal headaches.<br />

Socher et al.<br />

2. Hajiioannou J, Owens D, Whittet HB. Evaluation of<br />

anatomical variation of the crista galli using computed<br />

tomography. Clin Anat. 2010;23(4):370-3.<br />

3. Miranda CMNR, Maranhão COM, Arraes FMNR, Padilha<br />

IG, Farias LPG, Jatobá MAS, Andrade ACM, Padilha BG.<br />

Variações anatômicas das cavidades paranasais à tomografia<br />

computadorizada multislice: o que procurar? Radiol Bras.<br />

2011;44(4):256-62.<br />

4. Dutra LD, Marchiori E. Tomografia computadorizada<br />

helicoidal dos seios paranasais na criança: avalização das<br />

sinusopatias inflamatórias. Radiol Bras. 2002;35(3):161–9.<br />

5. Lee JM, Ransom E, Lee JYK, Palmer JN, Chiu AG.<br />

Endoscopic Anterior Skull Base Surgery: Intraoperative<br />

Considerations of the Crista Galli. Skull Base. 2011;21(2):83-<br />

6.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):251-256.<br />

DOI: 10.7162/S1809-97772013000300004<br />

Velopharyngeal dysfunction: a systematic review of major instrumental<br />

and auditory-perceptual assessments<br />

Lauren Medeiros Paniagua 1 , Alana Verza Signorini 2 , Sady Selaimen da Costa 3 , Marcus Vinicius Martins Collares 4 ,<br />

Sílvia Dornelles 5 .<br />

1) Speech Language Pathologist. Doctor of Science in Children’s and Teenager’s Health- Universidade Federal do Rio Grande do Sul-UFRGS. Professor of Speech-Language<br />

Pathology at Fatima College (RS).<br />

2) Undergraduate Speech Pathology Fellow - Universidade Federal do Rio Grande do Sul - UFRGS. Undergraduate fellow of CNPq.<br />

3) MD; MSc; PhD. Associate Professor - Department of Otolaryngology & Head and Neck Surgery/School of Medicine Universidade Federal do Rio Grande do Sul.<br />

4) MD; PhD. Associate Professor - Department of Surgery; Head, Plastic Surgery Section - School of Medicine /Universidade Federal do Rio Grande do Sul.<br />

5) Speech Pathology, MSc, PhD. Professor - Department of Speech Pathology/ Universidade Federal do Rio Grande do Sul.<br />

Institution: Universidade Federal do Rio Grande do Sul (UFRGS).<br />

Porto Alegre / RS - Brazil.<br />

Mailing address: Lauren Medeiros Paniagua - Universidade Federal do Rio Grande do Sul (UFRGS) - Avenida João Wallig <strong>17</strong>05/627 – Porto Alegre / RS - Brazil - Zip Code:<br />

91340-001 - E-mail: lmedeirospaniagua@yahoo.com.br<br />

Article received on December 2 nd , 2012. Article accepted on April 15 th , 2013.<br />

SUMMARY<br />

Introduction: Velopharyngeal dysfunction may cause impaired verbal communication skills in individuals with cleft lip and<br />

palate; thus, patients with this disorder need to undergo both instrumental and auditory-perceptual assessments.<br />

Objective: To investigate the main methods used to evaluate velopharyngeal function in individuals with cleft lip and palate<br />

and to determine whether there is an association between videonasoendoscopy results and auditory-perceptual assessments.<br />

Method: We conducted a systematic review of the literature on instrumental and auditory-perceptual assessments. We searched<br />

the PubMed, Medline, Lilacs, Cochrane, and SciELO databases from October to November 2012.<br />

Summary of findings: We found 1,300 studies about the topic of interest published between 1990 and 2012. Of these, 56 studies<br />

focused on velopharyngeal physiology; 29 studies presented data on velopharyngeal physiology using at least 1 instrumental<br />

assessment and/or 1 auditory-perceptual assessment, and 12 studies associated the results of both types of assessments. Only<br />

3 studies described in detail the analysis of both methods of evaluating velopharyngeal function; however, associations between<br />

these findings were not analyzed.<br />

Conclusion: We found few studies clearly addressing the criteria chosen to investigate velopharyngeal dysfunction and associations<br />

between videonasoendoscopy results and auditory-perceptual assessments.<br />

Keywords: Cleft Palate; Communication Disorders; Velopharyngeal Sphincter<br />

INTRODUCTION<br />

The velopharyngeal mechanism relies on the action<br />

of the velopharyngeal sphincter to control the distribution<br />

of voiced and voiceless airstream in both the oral cavity and<br />

the nasal cavity. Individuals with an impaired velopharyngeal<br />

mechanism will develop velopharyngeal dysfunction<br />

(VPD), which may compromise the verbal communication<br />

skills of patients with an intact velopharyngeal sphincter<br />

(VPS). One of the consequences of such dysfunction is<br />

hypernasality, which has a great impact on the individuals<br />

with this condition.<br />

Hypernasality induces nasal resonance in sounds<br />

that should not have this characteristic in articulate speech.<br />

This is caused by excessive nasal air emissions and weak<br />

intraoral pressure for some sounds. Nasal air escape and<br />

hypernasality are typical of VPD. Hypernasality is a<br />

resonance alteration that affects the emission of vowel<br />

sounds, whereas nasal air escape is a change in speech<br />

articulation that hinders the production of high pressure<br />

consonants such as plosives and fricatives (1).<br />

Several methods for evaluating the VPS have been<br />

designed. The choice of a specific evaluation method is<br />

directly related to the focus of interest of a clinical<br />

investigation and its need for accuracy. The use of 1<br />

auditory-perceptual assessment and at least 1 instrumental<br />

assessment is recommended for the analysis of<br />

velopharyngeal function (2, 3).<br />

Auditory-perceptual assessment is the main method<br />

for detecting possible changes in speech nasality, and<br />

provides data on the function of velopharyngeal structures<br />

during speech production. That is, this evaluation method<br />

makes it possible to detect specific symptoms of cleft<br />

palate that may or may not be associated with VPD (4, 5,<br />

6, 7). Because auditory-perceptual assessment is easily<br />

performed, it is the most commonly used evaluation<br />

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Paniagua et al.<br />

method in clinical practice (8, 9). Analysis of the test results<br />

has been widely discussed in the literature because of a<br />

lack of uniformity in the protocols used by researchers and<br />

institutions, which makes it difficult to compare study<br />

results (2).<br />

Videonasoendoscopy shows dynamic, direct, and<br />

natural images of the anatomical structures of the nasal<br />

cavity, pharynx, and larynx; this imaging method is thus<br />

one of the most appropriate tools for assessment of the VPS<br />

(10). Velopharyngeal closure patterns and the presence of<br />

a velopharyngeal gap (i.e., a residual opening during<br />

maximum contraction of the VPS) can be detected during<br />

performance of the test. Such patterns can also be identified<br />

during speech production, including the characteristics and<br />

degree of movement of the soft palate and pharyngeal<br />

walls (11, 12, 13).<br />

Based on both the literature and clinical practice, it<br />

is possible to state that the anatomy and physiology of the<br />

velopharyngeal mechanism are complex. Velopharyngeal<br />

dysfunctions that impair oral communication skills because<br />

of hypernasality, nasal air escape, and other disorders can<br />

be detected in individuals with cleft lip and palate. Therefore,<br />

the objective of the present review was to investigate the<br />

main methods used to evaluate the velopharyngeal function<br />

in individuals with cleft lip and palate, and to determine<br />

whether there is an association between<br />

videonasoendoscopy results and auditory-perceptual<br />

assessments.<br />

METHOD<br />

A systematic review of the literature is aimed at<br />

providing answers to specific questions and is based on<br />

clear and systematic methods in order to identify, select,<br />

and critically evaluate studies that may meet the proposed<br />

objective (14, 15). In order to perform the present systematic<br />

review of the literature, we searched studies conducted in<br />

different countries addressing the types of assessments<br />

used to describe the velopharyngeal function in individuals<br />

with surgically repaired cleft lip and palate. The following<br />

research questions were proposed: What are the main<br />

methods of velopharyngeal function assessment used in<br />

individuals with cleft lip and palate and how are the<br />

findings analyzed by the examiner? Is there an association<br />

between videonasoendoscopy results and auditoryperceptual<br />

assessments?<br />

We searched the PubMed, Medline, Lilacs, Cochrane,<br />

and SciELO databases from October to November 2012.<br />

The same search strategy was used for all databases. Our<br />

search strategy included only manuscripts published<br />

between 1990 and 2012. This period was selected based<br />

on the fact that videonasoendoscopy first appeared in the<br />

literature in 1990. First, we selected the keywords to search<br />

the databases considering our research questions. The<br />

following keywords were used alone and in combination<br />

with the other terms: “cleft palate,” “velopharyngeal closure,”<br />

“velopharyngeal insufficiency,” “velopharyngeal<br />

dysfunction,” “compensatory articulation,”<br />

“videonasoendoscopy,” “assessment and hypernasality,”<br />

“velopharyngeal mechanism,” “hypernasality,” and<br />

“speech.”<br />

The abstracts and titles of the manuscripts were<br />

selected by 2 researchers who worked independently. A<br />

reviewer resolved potential discrepancies of opinion. The<br />

full text of all potentially relevant manuscripts was obtained<br />

and analyzed separately by 2 reviewers based on the<br />

following inclusion criteria: (1) involved adults or children<br />

with cleft lip and palate; (2) included at least 1<br />

videonasoendoscopy and 1 auditory-perceptual assessment<br />

for screening of VPD; (3) described the methods and<br />

criteria used for the analysis of the velopharyngeal functional<br />

assessment results. Studies on cleft lip and palate focused<br />

on audiological findings, classification of different cleft<br />

types, and surgical interventions were excluded. Finally, 2<br />

researchers who specialize in the area of interest in the<br />

present study revised the selection of manuscripts with the<br />

purpose of refining the results.<br />

RESULTS AND DISCUSSION<br />

Considering the objective of the present review of<br />

the literature, our search of the previously mentioned<br />

scientific databases retrieved 1,300 manuscripts on<br />

velopharyngeal dysfunction in individuals with cleft lip and<br />

palate. Of these, 56 studies addressing velopharyngeal<br />

physiology were selected. Among these studies, there<br />

were 29 studies including data about auditory-perceptual<br />

assessment, some of which were associated with other<br />

instrumental assessments. We selected 12 studies that<br />

found an association between instrumental assessments<br />

(videonasoendoscopy and other tests) and auditoryperceptual<br />

assessment. Of these, 6 studies used<br />

videonasoendoscopy and auditory-perceptual assessment<br />

to evaluate velopharyngeal function. We refined our search<br />

in accordance with the objective of the present review and<br />

found only 3 studies that included an explanatory description<br />

of the analysis of both types of assessments.<br />

The characteristics of each study included in this<br />

review are shown in Table 1. The 12 studies selected were<br />

conducted in 4 different countries, including Brazil. These<br />

studies involved patients with different types of cleft lip<br />

and palate, including submucous cleft. Their age ranged<br />

from 3 to 76 years (Table 1).<br />

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Table 1. Characteristics of the study.<br />

Author Country Type of cleft Age group<br />

Araújo Netto & Cervantes, 2011 Brazil CLP, CP 4–19 years old<br />

Trindade et al., 2004 Brazil CLP, SCP NA<br />

Chanchareonsook et al., 2007 China CP 12–18 years old<br />

Kao et al., 2008 United States CP, SCP 7–11 years old<br />

Marsh, 2009 India CP, CLP NA<br />

Miguel et al., 2004 Brazil SCP, SCLP 6–46 years old<br />

Nagarajan et al., 2009 India CLP, CP NA<br />

Penido et al., 2007 Brazil CP 8–34 years old<br />

Qui Chen et al., 2011 United States SCP, CP 1–34 years old<br />

Camargo et al., 2001 Brazil NA 6–76 years old<br />

Shprintzen and Marrinan, 2009 United States NA NA<br />

Shyammohan et al., 2010 India NA NA<br />

Source: The authors<br />

CLP = cleft lip and palate<br />

CP = cleft palate<br />

SCP = submucuous cleft palate<br />

SCLP = submucous cleft lip and palate<br />

NA = not available<br />

Table 2. Tools used in the study.<br />

Authors Direct instrumental Indirect instrumental Clinical evaluation<br />

assessment<br />

assessment<br />

Araújo Netto & Cervantes, 2011 videonasoendoscopy Not used Auditory-perceptual assessmentl<br />

Trindade et al., 2004 Not used Not used Auditory-perceptual assessment<br />

Chanchareonsook et al., 2007 nasoendoscopy nasometry Auditory-perceptual assessment<br />

Kao et al., 2008 nasoendoscopy Not used Auditory-perceptual assessment<br />

Marsh, 2009 videonasoendoscopy Not used Auditory-perceptual assessment<br />

Miguel et al., 2004 videonasoendoscopy nasometry/PERCI-SARS Auditory-perceptual assessment<br />

Nagarajan et al., 2009 Videonasoendoscopy and nasometry Auditory-perceptual assessment<br />

videofluoroscopy<br />

Penido et al., 2007 nasopharyngoscopy Not used Nasal air emission test<br />

Qui Chen et al., 2011 lateral cephalogram of Not used Auditory-perceptual assessment<br />

nasopharyngography and<br />

nasopharyngeal fiberscope<br />

Camargo et al, 2001 nasoendoscopy Not used Auditory-perceptual assessment<br />

focused on nasal air emission<br />

Shprintzen e Marrinan, 2009 Nasopharyngoscopy and MRI nasometry Not used<br />

Shyammohan et al, 2010 Not used Not used Auditory-perceptual assessment<br />

Source: The authors.<br />

Detailed descriptions of both types of assessment<br />

and the parameters used for the analysis of the results of<br />

these 12 studies are shown in Table 2. The direct instrumental<br />

assessments used in these studies included<br />

videofluoroscopy, magnetic resonance imaging (MRI),<br />

nasopharyngeal fibroscopy, videonasoendoscopy,<br />

nasopharyngoscopy, and nasoendoscopy. Despite the<br />

different terms used, the last 4 assessments consisted of the<br />

same type of test performed with the purpose of viewing<br />

the velopharyngeal mechanism. The indirect instrumental<br />

assessments mentioned in these studies were nasometry<br />

and the PERCI-SARS system. The methods of clinical<br />

evaluation used in most studies were auditory-perceptual<br />

assessments and nasal airflow measurements. Such tests<br />

were analyzed according to the specific protocols of each<br />

institution. The analysis parameters for each type of<br />

instrumental assessment and auditory-perceptual<br />

assessment differed for each study (Table 2).<br />

Our decision to conduct a systematic review with<br />

the previously mentioned objective was prompted by a<br />

problem faced by health professionals who provide clinical<br />

care to patients with cleft lip and palate, that is, the impact<br />

of VPD on these patients’ verbal communication skills.<br />

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Discrepancies in findings relating to speech nasality and<br />

the degree of velopharyngeal closure are common in<br />

clinical practice. Clinical reports of verbal communication<br />

based on auditory-perceptual assessments reveal situations<br />

where the examiners detect severe hypernasality and,<br />

conversely, videonasoendoscopy shows a small<br />

velopharyngeal gap. Inversely proportional situations also<br />

occur when a large gap has little impact on nasality. Thus,<br />

the association between the clinical and instrumental<br />

evaluations is very important for achieving a clinical<br />

conclusion as to the real condition of the velopharyngeal<br />

mechanism. Even though we are aware that the<br />

pathophysiology of the velopharyngeal sphincter is<br />

complex and involves a variety of biases produced by the<br />

patient or the examiner, we searched for studies addressing<br />

this issue (i.e., the association between the size of the gap<br />

and the impact on nasality) in a straightforward manner.<br />

Therefore, the 3 studies selected are discussed further in an<br />

attempt to determine significant aspects related to the<br />

velopharyngeal mechanism and nasality. It is noteworthy<br />

that the specific association of interest in the present<br />

review was not explained by any of these studies, revealing<br />

the scarcity of scientific literature on the topic (Figure 1).<br />

While investigating this topic, Marsh (2009) addressed<br />

the velopharyngeal closure and provided a description of<br />

videonasoendoscopy and auditory-perceptual assessment<br />

in a study involving patients with cleft palate. Marsh suggested<br />

a classification of the residual gap in the VPS closure using<br />

different closure patterns during speech production, and<br />

considers investigation of the VPS closure pattern and<br />

analysis of lateral pharyngeal wall movement essential for<br />

establishing the diagnosis of VPD. Although there is no direct<br />

association between findings of nasality, audible nasal air<br />

emission, and the classification proposed, analysis of these<br />

findings does facilitate clinical surgical intervention.<br />

Qi Chen et al. (2011) used analysis of the lateral<br />

pharyngeal wall movement as a diagnostic criterion for<br />

VPS, considering that most patients without nasality and<br />

audible nasal air emission have sagittal closure. This study<br />

included 276 individuals aged 6 to 12 years. Despite<br />

considering factors such as surgical age and technique, Qi<br />

Chen et al. and Marsh both highlight the role played by<br />

lateral wall movement in satisfactory functioning of the<br />

VPS, and the consequences and impact on nasality.<br />

With the purpose of demonstrating an association<br />

between audible nasal air emission and VPS closure pattern<br />

shown by videonasoendoscopy, Penido et al. (2007) found<br />

a valid association between these aspects by means of<br />

comparison. These authors described the reliability of the<br />

correlation between the Glatzel mirror test using nasal air<br />

escape and VPS closure pattern in 21 individuals with cleft<br />

lip and palate whose mean age was <strong>17</strong> years. Based on<br />

Figure 1. Logistics associated with the systematic review.<br />

videonasoendoscopy, the size of the gap was categorized<br />

as small, medium, or large. This study demonstrated that<br />

the nasal air emission test is useful for evaluating the<br />

function of the velopharyngeal mechanism, and for<br />

establishing a direct relationship between the size of the<br />

gap and the area of condensation on the mirror. It is worth<br />

noting that these authors found divergent behaviors of the<br />

velopharyngeal mechanism considering the nasal air escape<br />

expected in an individual from this sample.<br />

CONCLUSIONS<br />

In the present systematic review of the literature we<br />

identified different evaluation methods used for determining<br />

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Paniagua et al.<br />

velopharyngeal function. However, we found few studies<br />

that showed both the detailed criteria chosen to analyze<br />

the results of the assessments and the association between<br />

videonasoendoscopy results and auditory-perceptual<br />

assessments. Only 1 study demonstrated an association<br />

between findings of VPD assessed using<br />

videonasoendoscopy and the severity of audible nasal air<br />

escape evaluated by auditory-perceptual assessment.<br />

REFERENCES<br />

1. Peterson-Falzone SJ, Hardin-Jones MA, Karnell MP.<br />

Communication disorders associated with cleft palate. In:<br />

Peterson-Falzone SJ, Hardin-Jones MA, Karnell MP, editors.<br />

Cleft Palate Speech. 3rd ed. Missouri: Mosby; 2001. p. 162-<br />

99.<br />

2. Rocha DL. Insuficiência Velofaríngea. In: Mélega JM.<br />

Cirurgia Plástica: Fundamentos e Arte - Cirurgia reparadora<br />

de Cabeça e Pescoço. Rio de Janeiro: Medsi; 2002. p. <strong>17</strong>8-<br />

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3. Golding-Kushner KJ, et al. Standartization for the reporting<br />

of nasopharyngoscopy and multiview videofluoroscopy: a<br />

report from an international working group. Cleft Palate J.<br />

1990;27:337-48.<br />

4. Conley SF, Gosain AK, Marks SM, Larson DL. Identification<br />

and assessment of velopharyngeal inadequacy. Am J<br />

Otolaryngol. 1997;18:38-46.<br />

5. Shprintzen RJ. Instrumental Assessment Of<br />

Velopharyngeal Valving. In: Shprintzen RJ, Bardach J. Cleft<br />

palate speech management: A multidisciplinary approach.<br />

St. Louis: Mosby; 1995. p. 221-56.<br />

6. Trindade IEK, Trindade Junior AS. Avaliação funcional da<br />

inadequação velofaríngea. In: Carreirão S, Lessa S, Zanini<br />

AS, editors. Tratamento das fissuras labiopalatinas. 2nd ed.<br />

Rio de Janeiro: Editora Revinter; 1996. p. 223-35.<br />

7. Sell D, Harding A, Grunwell P. A screening assessment<br />

of cleft palate speech (Great Ormond Street Speech<br />

Assesmente). Eur J Disord Commun. 1999;29:1-15.<br />

8. Genaro KF, Yamashita RP, Trindade IKE. Avaliação clínica<br />

e instrumental na fissura labiopalatina. In: Ferreira LP, Befi-<br />

Lopes DM, Limongi SCO. Tratado de Fonoaudiologia. São<br />

Paulo: Rocca; 2004. p. 456-77.<br />

9. Laczi E, Sussman JE, Stathopoulos ET, Huber J. Perceptual<br />

evaluation of hypernasality compared to HONC measures:<br />

The role of experience. Cleft Palate Craniofac J. 2005;42:202-<br />

10.<br />

10. Pontes PAL, Behlau MS. Nasolaringoscopia. In: Altmann<br />

EBC. Fissuras labiopalatinas. 4th ed. Carapicuíba: Pró-fono;<br />

2005. p. <strong>17</strong>5-83.<br />

11. Kuehn DP, Henne LJ. Speech Evaluation and Treatment<br />

for Patients With Cleft Palate. Am J Speech Lang Pathol.<br />

2003;12:103–9.<br />

12. Shprintzen RJ. Nasopharyngoscopy. In: Bzoch KR,<br />

editors. Communicative Disorders Related To Cleft Lip And<br />

Palate. 5th ed. Boston: Little & Brown; 2004.<br />

13. Williams WN, Heningsson G, Pegoraro-Krook MI.<br />

Radiographic assessment of Velopharyngeal function for<br />

speech. In: Bzoch KR, editor. Communicative disorders<br />

related to cleft lip and palate. 5th ed. Boston: Little & Brown;<br />

2004.<br />

14. Klassen AF, Tsangaris E, Forrest CR, Wong KWY, Pusic<br />

AL, Cano SJ, et al. Quality of life of children treated for cleft<br />

lip and/or palate: A systematic review. J Plast Reconstr<br />

Aesthet Surg. 2012;65:547-57.<br />

15. Sampaio RF, Mancini MC. Estudos De Revisão Sistemática:<br />

Um Guia Para Síntese Criteriosa Da Evidência Científica.<br />

Rev. Bras. Fisioter. 2007;11:83-9.<br />

16. Camargo LOS, Rodrigues MC, Avelar JA. Oclusão<br />

velofaríngea em indivíduos submetidos à nasoendoscopia<br />

na Clínica de Educação para Saúde (CEPS). Salusvita.<br />

2001;20:35-47.<br />

<strong>17</strong>. Chanchareonsook N, Whitehill TL, Samman N. Speech<br />

outcome and velopharyngeal function in cleft palate:<br />

comparison of Le Fort I maxillary osteotomy and distraction<br />

osteogenesis—early results. Cleft Palate Craniofac J.<br />

2007;44:23-32.<br />

18. Kao DS, Soltysik DA, Hyde JS, Gosain AK. Magnetic<br />

Resonance Imaging as an Aid in the Dynamic Assessment<br />

of the Velopharyngeal Mechanism in Children. Plast Reconstr<br />

Surg. 2008;122:572–7.<br />

19. Marsh J. Velo-pharyngeal dysfunction: Evaluation and<br />

management. Indian J Plast Surg. 2009;42:129–36.<br />

20. Miguel HC, Genaro KF, Trindade IEK. Avaliação<br />

perceptiva e instrumental da função velofaríngea na fisura<br />

de palato submucosa assintomática. Pró-Fono <strong>Revista</strong> de<br />

Atualização Científica. 2007;19:105-12.<br />

21. Nagarajan R, Savitha VH, Subramaniyan B.<br />

Communication disorders in individuals with cleft lip and<br />

palate: An overview. Indian J Plast Surg. 2009;42:137–<br />

43.<br />

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22. Netto BCA, Cervantes O. Estudo comparativo entre<br />

pacientes fissurados portadores de insuficiência velofaríngea<br />

tratados com fonoterapia e faringoplastia. Rev. Bras. Cir.<br />

Plást. 2011;26:631-8.<br />

23. Penido FA, Noronha RMS, Caetano KI, Jesus MSV, Di<br />

Ninno CQMS, Britto ATBO. Correlação entre os achados do<br />

teste de emissão de ar nasal e da nasofaringoscopia em<br />

pacientes com fissura labiopalatina operada. Rev Soc Bras<br />

Fonoaudiol. 2007;12:126-34.<br />

24. Shprintzen RJ, Marrinan EMS. Velopharyngeal<br />

insufficiency: diagnosis and management. Curr Opin<br />

Otolaryngol Head Neck Surg. 2009;<strong>17</strong>:302-7.<br />

25. Shyammohan A, Sreenivasulu D. Speech Therapy with<br />

Obturator. J Indian Prosthodont Soc. 2010;10:197–9.<br />

26. Trindade IEK, Genaro KF, Yamashita RP, Miguel HC,<br />

Fukushiro AP. Proposta de classificação da função<br />

velofaríngea na avaliação perceptivo-auditiva da fala. Pró-<br />

Fono <strong>Revista</strong> de Atualização Científica. 2005;<strong>17</strong>:259-62.<br />

27. Qi C, Qian Z, Bing S, Heng Y, Tian M, Guang-ning Z.<br />

Study of relationship between clinical factors and<br />

velopharyngeal closure in cleft palate patients. J Res Med<br />

Sci. 2011;16:945–50.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):257-264.<br />

DOI: 10.7162/S1809-97772013000300005<br />

Results obtained with a low cost software-based audiometer for hearing<br />

screening<br />

Deborah Viviane Ferrari 1 , Esteban Alejandro Lopez 2 , Andrea Cintra Lopes 3 , Camila Piccini Aiello 4 , Pricila Reis Jokura 5 .<br />

1) PhD in Neuroscience and Behavior - Institute of Psychology, University of São Paulo. Professor, Department of Speech Language Pathology and Audiology. Dentistry<br />

School of Bauru - University of São Paulo.<br />

2) Clinical Engineer. Master in Biomedical Engineering - Favaloro University, Buenos Aires, Argentina. Favaloro University, Buenos Aires, Argentina.<br />

3) PhD in Communication Disorders - University of São Paulo. Associate Professor, Department of Speech Language Pathology and Audiology. Dentistry School of Bauru<br />

- University of São Paulo.<br />

4) Speech Language Pathologist and Audiologist. Graduate Student - Master in Speech Language Pathology and Audiology. Dentistry School of Bauru - University of<br />

São Paulo.<br />

5) Speech Language Pathologist and Audiologist. Graduate Student - Master in Speech Language Pathology and Audiology. Dentistry School of Bauru - University of<br />

São Paulo.<br />

Institution: Departamento de Fonoaudiologia. Faculdade de Odontologia de Bauru - USP.<br />

Bauru / SP - Brazil.<br />

Mailing address: Faculdade de Odontologia de Bauru – USP. Deborah Viviane Ferrari Al. Octávio Pinheiro Brisola 9-75. Vila Universitária - Bauru / SP – Brazil - Zip code:<br />

<strong>17</strong>012-901. E-mail: deborahferrari@usp.br<br />

Article received on December 10 th , 2012. Article accepted on April 7 th , 2013.<br />

SUMMARY<br />

Introduction: The implementation of hearing screening programs can be facilitated by reducing operating costs, including the<br />

cost of equipment. The Telessaúde (TS) audiometer is a low-cost, software-based, and easy-to-use piece of equipment for<br />

conducting audiometric screening.<br />

Aim: To evaluate the TS audiometer for conducting audiometric screening.<br />

Methods: A prospective randomized study was performed. Sixty subjects, divided into those who did not have (group A, n<br />

= 30) and those who had otologic complaints (group B, n = 30), underwent audiometric screening with conventional and TS<br />

audiometers in a randomized order. Pure tones at 25 dB HL were presented at frequencies of 500, 1000, 2000, and 4000 Hz.<br />

A “fail” result was considered when the individual failed to respond to at least one of the stimuli. Pure-tone audiometry was<br />

also performed on all participants. The concordance of the results of screening with both audiometers was evaluated. The<br />

sensitivity, specificity, and positive and negative predictive values of screening with the TS audiometer were calculated.<br />

Results: For group A, 100% of the ears tested passed the screening. For group B, “pass” results were obtained in 34.2% (TS)<br />

and 38.3% (conventional) of the ears tested. The agreement between procedures (TS vs. conventional) ranged from 93% to 98%.<br />

For group B, screening with the TS audiometer showed 95.5% sensitivity, 90.4% sensitivity, and positive and negative predictive<br />

values equal to 94.9% and 91.5%, respectively.<br />

Conclusions: The results of the TS audiometer were similar to those obtained with the conventional audiometer, indicating<br />

that the TS audiometer can be used for audiometric screening.<br />

Keywords: Hearing; Hearing Loss; Hearing Tests.<br />

INTRODUCTION<br />

Hearing loss is among the three most prevalent<br />

conditions worldwide, with around 636.5 million people<br />

suffering from a certain degree of hearing loss (1). It is<br />

estimated that in Brazil, 6.8% of the population suffers from<br />

disabling hearing loss, i.e., a hearing threshold level for the<br />

better ear of 41 dB HL or greater, averaged at frequencies<br />

of 0.5, 1, 2, and 4 kHz. Of these individuals, 5.3% are<br />

children between 4 and 9 years old, 36.2% are elderly, and<br />

19.5% are adults between 20 and 59 years old (2). In the<br />

state of São Paulo, a population-based study showed that<br />

the prevalence of hearing loss was 44%, being higher for<br />

elderly male individuals (3). A population-based crosssectional<br />

study performed in Rio de Janeiro showed that,<br />

for the elderly, the prevalence of hearing loss in the better<br />

ear was 42.9% (4).<br />

Hearing loss can hinder or even impede acquisition<br />

and development of oral language, as well as academic and<br />

social development in children. In addition, it can decrease<br />

the quality of life and work and educational opportunities of<br />

adult individuals. This condition also has an impact on society<br />

in terms of loss of productivity of affected individuals as well<br />

as costs of treatment, education, and rehabilitation of people<br />

with special needs. Thus, healthcare actions focused on<br />

prevention, early identification, and treatment of hearing<br />

problems also have social and economic implications.<br />

The Brazilian National Health System (Sistema Único<br />

de Saúde, SUS) has incorporated the treatment of the<br />

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hearing impaired in previous decades. Such action was<br />

supplemented by the establishment of the National Hearing<br />

Healthcare Policy in 2004 (5) and, more recently, by the<br />

Care Network for People with Disabilities within the SUS<br />

(6). This network is <strong>org</strong>anized into the components of<br />

primary care, specialized rehabilitation care, and emergency<br />

hospital care. Primary care services are provided through<br />

Healthcare Basic Units (UBS), which prioritize certain<br />

strategic actions intended to enhance access for and<br />

attention to people with disabilities, including the promotion<br />

of early identification services.<br />

In Brazil, newborn universal hearing screening has<br />

been enforced by law since 2010 (7). However, progressive<br />

or late onset hearing losses are not detected at birth. It is<br />

estimated that 9–10 out of 1000 school aged children have<br />

permanent hearing loss in at least one ear (8). The Health<br />

in School Program (PSE), initiated in 2007 as a joint action<br />

between the Ministries of Health and Education, aims to<br />

incorporate the school community into projects and<br />

programs that make use of healthcare and education in<br />

order to comprehensively deal with the issues that<br />

compromise the development of Brazilian children and<br />

teenagers. The primary task of the PSE is the assessment<br />

of the health condition of students, including hearing status.<br />

In order to do so, audiometric procedures, among others,<br />

are recommended (9).<br />

For adults and the elderly, hearing loss onset is often<br />

insidious, complicating self-perception of the problem<br />

and, consequently, the request for treatment. Performing<br />

hearing screening in this population would enable timely<br />

identification and corresponding referral to rehabilitation<br />

services, in order to minimize secondary consequences<br />

such as functional decline, depression, and social isolation.<br />

In Brazil, there are still no large scale hearing<br />

screening programs targeting school aged children, adults,<br />

and the elderly. When screenings are conducted in this<br />

population, they are isolated events. The reasons for this<br />

scenario are multifactorial and include the costs of the<br />

procedure. The related costs can be attributed to the cost<br />

of the professional performing the procedure, the necessary<br />

time to perform it, and the cost of the necessary equipment.<br />

In order to decrease screening costs, a simple, fast, and<br />

effective method using low cost equipment that can be<br />

easily operated by a primary care professional must be<br />

utilized (10).<br />

It is also necessary to emphasize the difficulty of<br />

access to hearing health for populations living far from<br />

urban centers. Thus, a relevant aspect of this scenario is the<br />

development of computer-based systems that can be used<br />

in telehealth models. Different low cost systems for<br />

conducting audiometric screening (11-13) or audiological<br />

assessment at distance (14-15) have been published in the<br />

literature. Such systems use TDH-39 supra-aural headphones<br />

or ER3A insert phones, which can increase device cost.<br />

One study was found on the use of a software-based<br />

screening audiometer that utilized a conventional<br />

circumaural phone with a 3.5 mm plug. However, this<br />

system was automatic and hearing responses were queried<br />

with 3 dB increments, making the test relatively lengthy<br />

with a duration of about 15 minutes (10).<br />

The Telessaúde (TS) audiometer was developed to<br />

perform audiometric screening using off-the-shelf USB<br />

interface headphones. This characteristic has a relevant<br />

impact on acquisition and replacement costs (the prototype<br />

costs less than 50 USD) and also facilitates access of the<br />

user to the TS audiometer. A wide variety of USB headphones<br />

are available and the user can chose a model that is<br />

considered more convenient, once that set of headphones<br />

has been calibrated for use with the TS audiometer. The<br />

fact that only off-the-shelf electronic devices are used also<br />

facilitates the availability of the TS audiometer, as the<br />

eventual manufacturer will not require a specific<br />

infrastructure for manufacturing and distribution. The aim<br />

of this study was to evaluate the TS audiometer for use<br />

during audiometric screening.<br />

METHOD<br />

This randomized prospective study was conducted<br />

in the Speech Language Pathology and Audiology Clinic of<br />

the X School, University X, and was approved by the<br />

relevant research ethics committee (process number 021/<br />

2010). This clinic is currently accredited by the SUS as a<br />

tertiary hearing healthcare service.<br />

Once an informed consent form was signed by 60<br />

individuals, aged 18 years or older, they were voluntarily<br />

enrolled in the study and segregated into 2 groups (Table<br />

1):<br />

• Group A: for this group, employees and graduate<br />

students of the Bauru School of Dentistry were invited<br />

to participate in the study. This group was composed<br />

of 30 adults with age varying from 18 to 41 years old.<br />

None of these individuals presented a hearing complaint.<br />

• Group B: for this group, individuals who had ENT or<br />

audiological complaints and were referred to the clinic<br />

were invited to participate in the study. This group was<br />

comprised of 30 individuals with age ranging from 23<br />

to 85 years old. There were 12 adults and 18 elderly<br />

subjects (aged 60 years or older). None of these<br />

individual had had their hearing assessed previously.<br />

The TS audiometer is integrated by software<br />

developed in Visual Basic.NET 2005 (Net Framework 2.0)<br />

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Table 1. Demographic data of the participants.<br />

Group Number of Age in years Gender Complaint<br />

ears tested (mean ± SD)<br />

Male Female Tinnitus Dizziness Hearing difficulty<br />

A (n = 30) 60 23.2 ± 5.6 26 4 —- —- —-<br />

B (n = 30) 60 59 ± 18.2 16 14 11 3 18<br />

for Windows XP and a set of USB headphones. It is<br />

supported by a multiuser database system managed by an<br />

administrator. The system can store the registration<br />

information of the location where screening is performed,<br />

the information of the healthcare professionals (users) and<br />

screened populations, as well as the corresponding<br />

evaluation results. Figure 1 shows an example of a TS<br />

audiometer user interface.<br />

With regards to hardware, the audiometer uses a<br />

Microsoft LifeChat LX-3000 headset with the following<br />

specifications: (a) bilateral earphones (20 Hz–20 kHz); (b)<br />

embedded microphone (100 Hz–10 kHz); (c) embedded<br />

USB sound board with 16 bit precision; (d) plug and play,<br />

i.e., no other software installation is needed for normal use;<br />

and (e) incorporated volume control. As for every USB<br />

device, the headset is identified by the host computer by<br />

its Vendor ID and Product ID, therefore allowing the<br />

software to apply the corresponding calibration parameter<br />

or impede the performance of the audiometric procedure<br />

if the corresponding parameters are not available.<br />

Following FDA recommendations for signature of<br />

electronic records (16), the identity and electronic signature<br />

of the healthcare professional are protected by a password<br />

only known and modifiable by the healthcare professional.<br />

Additionally, a change control, or audit trail, is not deemed<br />

necessary, as once they are electronically signed, the<br />

assessment results can no longer be modified.<br />

In this study, the TS audiometer was installed and<br />

tested in an ASUS EeePc900 laptop, given its relatively<br />

low cost and portability. This laptop has a 8.9" screen, a<br />

Celeron M353 processor, a 4 GB hard drive, 1 GB of DDR<br />

II RAM, an Intel UMA video board, a 1.3 megapixel<br />

webcam, a Windows ® XP operating system, 802.11b/g<br />

WLAN, USB/VGA/Earphone/Mic/Network inputs/outputs,<br />

embedded loudspeakers, a battery autonomy of<br />

approximately 2.5 hours, dimensions of 22.5x<strong>17</strong>x2 cm,<br />

and a weight of 0.99 kg.<br />

The TS audiometer includes a calibration user<br />

interface. This interface is only accessible for the testing<br />

prototype. Through this interface, the calibration parameters<br />

of a certain headset model can be determined and stored.<br />

These values are stored in the system parameters database<br />

Figure 1. TS audiometer user interface.<br />

and they are used every time an audiometric screening is<br />

performed. The device (computer and headphones) was<br />

calibrated by an engineer with experience in conventional<br />

audiometer calibration according to the applicable<br />

requirements of the standard project ABNT/CB 03/CE-<br />

03:029.01-022/1. The frequencies 250–8000 Hz were<br />

used to calibrate the left and right earphones of the<br />

Microsoft LifeChat LX-3000 headset. During calibration, the<br />

TS audiometer software was used to provide acoustic<br />

stimulation. With regard to the calibration, it must be noted<br />

that the USB headset included its own sound board, i.e., its<br />

own analog and digital input/output stages. Therefore, the<br />

calibration parameters were characteristic of the headset<br />

model and independent of the host computer model or<br />

type. The frequencies 250, 500, 1000, 2000, 3000, 4000,<br />

6000, and 8000 Hz were calibrated ranging from 10 dB HL<br />

(minimum stimulation level) to 70 dB HL (maximum<br />

stimulation level) in 5 dB steps for both the right and left<br />

channels.<br />

Another feature contemplated during development<br />

of the TS audiometer was the real-time estimation of<br />

ambient noise levels during performance of the screening<br />

procedure. As audiometric screening is not necessarily<br />

conducted in an audiometric booth or an acoustically prepared<br />

room, the TS audiometer uses the microphone embedded<br />

in the headset, placed in the upright position, to determine<br />

the ambient noise level. The software application converts<br />

the sampled noise level into a dB scale in order to assess if<br />

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the real-time ambient noise level allows screening to be<br />

performed. The system will automatically display a message<br />

to the evaluator when the ambient noise level is greater than<br />

60 dB SPL to make him/her aware that minimization of noise<br />

level is desirable. The system user interface will flag the<br />

responses obtained under an ambient noise level greater<br />

than 60 dB SPL and also stores the average ambient noise<br />

level measured during an evaluation on its database. This is<br />

intended to minimize the occurrence of false positives<br />

induced by the lack of an appropriate acoustic environment.<br />

As the screenings were conducted in a sound booth, this<br />

feature of the TS audiometer was not evaluated in the<br />

present study.<br />

Audiometric screening was conducted in groups A<br />

and B with 2 different pieces of equipment: the SD 50<br />

audiometer (Siemens) coupled with supra-aural THD-39<br />

headphones and the TS audiometer. The screenings were<br />

conducted on the same day by 2 different audiologists,<br />

each one operating one audiometer. The audiologists did<br />

not share the results obtained with each other and they<br />

were not aware of the patients’ hearing complaints. The<br />

procedures were conducted in a randomized order.<br />

Audiometric screening was based on the ASHA<br />

protocol <strong>17</strong> . Pure tones at frequencies of 500, 1000, 2000,<br />

and 4000 Hz were presented at a 25 dB HL level. The<br />

participant was instructed to raise his/her hand each time<br />

the acoustic stimuli were heard. In the audiometric screening,<br />

a “pass” result was considered when the subject responded<br />

to the stimuli presented. The result was considered “fail”<br />

when the subject did not respond to one or more stimuli<br />

presented in one or both ears.<br />

Following audiometric screening, and regardless of<br />

the result (pass or fail), all participants underwent otologic<br />

inspection, pure-tone threshold audiometry, and speech<br />

audiometry procedures. For such purposes, SD 50<br />

(Siemens) or Midimate 622 (Madsen) audiometers were<br />

used. Air conduction hearing thresholds were obtained<br />

with TDH-39 earphones for the inter-octaves of the<br />

frequencies between 250 and 8000 Hz, for both ears.<br />

When the air conduction hearing thresholds were greater<br />

than 20 dB HL, bone conduction audiometry was also<br />

performed for the frequencies 500, 1000, 2000, 3000, and<br />

4000 Hz.<br />

Hearing thresholds were determined by applying<br />

the ascendant-descendant strategy. At each pure-tone<br />

detection response, the presentation level was reduced by<br />

10 dB until the individual no longer responded to the<br />

stimuli. Then, the presentation level was increased in 5 dB<br />

steps until a response was detected. The hearing threshold,<br />

at each frequency, was the lowest level at which the<br />

individual could detect 50% of the stimuli presented.<br />

All procedures were performed in a sound booth,<br />

where the noise levels were within the specified ranges of<br />

the ANSI 1999 standard (18).<br />

Concordance analysis and Cohen’s kappa coefficient<br />

were used to analyze the screening results between the<br />

conventional and TS audiometers for groups A and B.<br />

The precision of the TS audiometer screening<br />

instrument was evaluated through specificity, sensitivity,<br />

positive predictive value, and negative predictive value<br />

analysis. The sensitivity was defined as the percentage of<br />

ears that failed screening with the TS audiometer among<br />

those in which hearing loss was observed with pure-tone<br />

threshold audiometry. The specificity was defined as the<br />

percentage of ears that passed screening among those<br />

with normal hearing results. In this study, air conduction<br />

audiometric thresholds less than or equal to 20 dB HL were<br />

considered as normal hearing. The positive and negative<br />

predictive values were defined as the probability of a<br />

patient having hearing loss if they failed screening and the<br />

probability of a patient having normal hearing if they<br />

passing screening, respectively.<br />

RESULTS<br />

With regard to group B, the number of individuals<br />

that failed the screening was 27 with the TS audiometer<br />

and 26 with the conventional audiometer.<br />

Within group B, 4 participants presented normal<br />

hearing at all frequencies. The other 26 participants<br />

presented different degrees of sensorineural hearing loss<br />

(Figure 2), as per the pure-tone and speech audiometry<br />

results.<br />

Figure 2. Mean and standard deviation of audiometric thresholds<br />

for participants who presented hearing loss (n = 26).<br />

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Table 2. Audiometric screening and concordance results for group A (n = 60 ears).<br />

Frequency (Hz) TS audiometer Conventional audiometer Concordance (%)<br />

Pass Fail Pass Fail<br />

500 60 0 60 0 100<br />

1000 60 0 60 0 100<br />

2000 60 0 60 0 100<br />

4000 60 0 60 0 100<br />

Total (%) 240 (100) 0 (100) 240 (100) 0 (100) 100<br />

Table 3. Audiometric screening and concordance results for group B (n = 60 ears).<br />

Frequency (Hz) TS audiometer Conventional audiometer Concordance (%) Kappa coefficient<br />

Pass Fail Pass Fail<br />

500 32 28 32 28 98.31 0.97<br />

1000 24 36 28 32 93.33 0.86<br />

2000 20 40 22 38 96.67 0.93<br />

4000 06 54 10 50 96.33 0.71<br />

Total (%) 82 (34.2) 158 (65.8) 92 (38.3) 148 (61.4) 96.67 0.84<br />

DISCUSION<br />

In group A (Table 2), all tested ears passed the<br />

screening. Although the sample size was limited, this<br />

observation is supported by the fact that the participants<br />

were predominantly young females, a population for<br />

which the prevalence of hearing loss is smaller (2-3). The<br />

concordance between the screening results obtained with<br />

the conventional and TS audiometers was excellent.<br />

In group B (Table 3), most ears failed the screening<br />

with both audiometers. It is worth noting that this group of<br />

participants had some kind of otologic complaint, with 18<br />

of them reporting hearing difficulties. This group was also<br />

mostly composed of elderly subjects, for whom a greater<br />

prevalence of hearing loss is observed (2-4). Thus, a<br />

considerable number of failures were expected.<br />

The number of failures at the frequency of 500 Hz<br />

was less than that for the other frequencies, which contradicts<br />

results reported in the literature (10). This can be explained<br />

by the fact that the screening was conducted in a sound<br />

booth, this being a limitation for the generalization of the<br />

data from the current study. In fact, hearing screening is<br />

generally conducted in a non-acoustically isolated room.<br />

Consequently, ambient noise can exert a masking effect<br />

over emitted signals, which is more significant at low<br />

frequencies, thus affecting screening results at these<br />

frequencies. For this reason, several screening protocols<br />

exclude the 500 Hz test, although this frequency is<br />

relevant for the assessment of the impact of middle ear<br />

condition on hearing sensitivity.<br />

In Table 3, the kappa coefficients show an excellent<br />

concordance between the evaluators for the screening<br />

results obtained with the conventional and TS audiometers<br />

at the frequencies of 500 Hz to 3000 Hz, and substantial<br />

concordance for the frequency of 4000 Hz (19). The interevaluator<br />

reliability allows verification of the degree of<br />

correspondence between the independent evaluations of<br />

2 or more evaluators classifying the same phenomena.<br />

Therefore, it is a relevant indicator of the quality of the<br />

screening procedure with the TS audiometer. Previously,<br />

kappa coefficients of 0.79–0.93 were observed when<br />

evaluating the concordance between audiometric<br />

screenings conducted with a portable audiometer and a<br />

conventional audiometer 20 . Another study 10 indicated a<br />

kappa coefficient of 0.20 between screening performed<br />

with a software-based audiometer and a conventional one.<br />

The fact that this result is lower than the one obtained in<br />

the current study could be related to the population<br />

characteristics (school aged children) and the impact of<br />

ambient noise at the frequency of 500 Hz, since when this<br />

was excluded from the analysis, a kappa value of 0.62 was<br />

obtained. Another important difference is that an automatic<br />

screening procedure was employed in the previous study,<br />

in contrast with the conventional procedure utilized in the<br />

current study.<br />

In Table 4, it can be observed that audiometric<br />

thresholds for group A were within normal values.<br />

Audiometric thresholds greater than 25 dB HL were found<br />

for at least one frequency in 65.4% of the ears tested in<br />

group B. For this group, 26 participants (86.6%) presented<br />

sensorineural hearing loss, with a greater involvement of<br />

the high frequencies (Figure 2). This high incidence of<br />

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Table 4. Distribution of the number of ears with normal thresholds and<br />

hearing loss in both groups.<br />

Frequency (Hz) Group A (n = 60 ears) Group B (n = 60 ears)<br />

Normal Hearing loss Normal Hearing loss<br />

500 60 0 29 31<br />

1000 60 0 29 31<br />

2000 60 0 19 41<br />

4000 60 0 6 54<br />

Total (%) 240 (100) 0 (0) 83 (34.6) 157 (65.4)<br />

Table 5. Specificity, sensitivity, positive predictive value, and negative predictive<br />

value for TS and conventional audiometers.<br />

Audiometer Group Predictive value<br />

Sensitivity Specificity Positive Negative<br />

TS A —— 100% —— 100%<br />

B 95.5% 90.4% 94.9% 91.5%<br />

Conventional A —— 100% —— 100%<br />

B 91.1% 94% 96.6% 84.8%<br />

hearing loss is due to the fact that the participants of this<br />

group were recruited from a specialized hearing healthcare<br />

clinic. It is relevant to note that all of the subjects who<br />

presented hearing loss underwent a complete audiological<br />

assessment and received corresponding treatment, including<br />

fitting of hearing aids.<br />

Pure-tone audiometry is currently the gold standard<br />

for assessment of hearing sensitivity. Therefore, the<br />

sensitivity and specificity of the screening procedures<br />

with the conventional and TS audiometers (Table 5) were<br />

calculated using the results obtained with pure-tone<br />

threshold audiometry as a reference. It was not possible<br />

to calculate the sensitivity of the procedures for group A<br />

due to the fact that none of the participants failed the<br />

screening or presented hearing loss with pure-tone<br />

audiometry. The specificity of this group equaled 100%<br />

for both procedures. For group B, the sensitivity and<br />

specificity values of the TS audiometer were similar to<br />

those of the conventional audiometer (Table 5). Elevated<br />

sensitivity and specificity values avoid the occurrence of<br />

false negatives and false positives, respectively. However,<br />

it must be remembered that no procedure is entirely<br />

accurate, i.e., no procedure has a sensitivity and specificity<br />

equal to 100%.<br />

The sensitivity and specificity values of the TS<br />

audiometer were similar to those found in the literature.<br />

The sensitivity and specificity of a portable screening<br />

audiometer were reported to be 91.8–98.5% and 88.0–<br />

96.3%, respectively (20). Studies involving the Audioscope<br />

device with pure-tone sweep (500 to 4000 Hz) for hearing<br />

screening demonstrated a sensitivity of 94–97% and a<br />

specificity of 69–80% (11). With regards to affordable<br />

audiometers, the TS audiometer presented higher sensitivity<br />

and specificity values than those found in the literature.<br />

Sensitivity and specificity values of 86.7% and 75.9% were<br />

verified with a remote automatic audiometric screening<br />

method (20 dB HL pure-tone sweep), based on the use of<br />

a computer and TDH-39 earphones (13). Automatic hearing<br />

screening conducted with a low cost audiometer and a<br />

circumaural phone showed a sensitivity of 100% and a<br />

specificity of 49% (10). The differences observed between<br />

the literature and the present study can be related to the<br />

stimuli loudness level applied in the current study (25 dB<br />

HL), attenuation differences in the earphones utilized, and,<br />

mainly, the screening method (automatic vs. manual) and<br />

screening room acoustics (non-acoustically isolated room<br />

vs. audiometric booth) used.<br />

Under operational conditions (field application), the<br />

performance indicators of a test procedure are modified by<br />

the frequency of occurrence of a medical condition within<br />

the population (prevalence). Therefore, the predictive<br />

value of the procedure has a significant relevance, i.e., the<br />

probability of occurrence of a medical condition given a<br />

positive or negative result. In the current study, the<br />

positive and negative predictive values of the TS audiometer<br />

were equal to 94.9% and 91.5%, respectively, being similar<br />

to those obtained with a conventional audiometer (Table<br />

5). This means that if a subject fails screening with the TS<br />

audiometer, there is a 94.9% chance of them actually<br />

suffering from hearing loss and a 5.1% (100 - 94.9) chance<br />

of them having normal hearing. If the subject passes<br />

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Ferrari et al.<br />

screening, there is 91.5% chance of them having normal<br />

hearing and an 8.5% (100 - 91.5) chance of them having<br />

hearing loss.<br />

For group B, the observed predictive values of the<br />

TS audiometer were greater than those found in the<br />

literature for other affordable audiometers, which<br />

presented positive and negative predictive values of<br />

48.1% and 95.7% 13 and 56% and 43% 10 , respectively.<br />

These findings are justified by the differences observed<br />

in the sensitivity and specificity of the TS audiometer<br />

when compared to other audiometers, and the fact that<br />

the screened population was from a specialized hearing<br />

healthcare clinic, where a higher number of subjects with<br />

hearing loss were registered.<br />

Screening programs are justified if there is evidence<br />

that sustains 3 standard criteria: (a) negative consequences<br />

of the medical condition must be sufficiently significant in<br />

order to justify the screening effort; (b) an effective<br />

treatment for the detected medical condition is available;<br />

and (c) an accurate, practical, and convenient screening<br />

test is available 11 . Although this study is limited by the<br />

number of participants, the results obtained suggest that<br />

the TS audiometer meets the necessary criteria for a<br />

screening procedure.<br />

CONCLUSION<br />

The specificity, sensitivity, and positive and negative<br />

predictive values of audiometric screening conducted with<br />

the TS audiometer were high and similar to those obtained<br />

with a conventional audiometer.<br />

ACKNOWLEDGEMENTS<br />

The authors would like to thank Audiologist X for her<br />

support during data collection and Engineer X for performing<br />

the calibration of the TS audiometer.<br />

REFERENCES<br />

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disease/GBD_report_2004update_ part3.pdf<br />

2. Béria JU, Raymann BCW, Gigante LP, Figueiredo AL, Jotz<br />

GP, Roithmann R, et al. Hearing impairment and<br />

socioeconomic factors: a population-based survey of an<br />

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Public Health. 2007;21:381-7.<br />

3. Castro SS, César CLG, Carandina L, Barros MBA, Alves<br />

MCGP, Goldbaum M. Deficiência visual, auditiva e física:<br />

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4. Mattos LC, Veras RP. Prevalência da perda auditiva em<br />

uma população de idosos da cidade do Rio de Janeiro: um<br />

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2007;73(5):654-9 .<br />

5. BRASIL, Ministério da Saúde. Portaria n° 2.073 de 28 de<br />

setembro de 2004. Institui a política nacional de saúde<br />

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/dtr2001.saude.gov.br/sas/PORTARIAS/Port2004/GM/GM-<br />

2073.htm<br />

6. BRASIL, Ministério da Saúde. Portaria n° 793 de 24 de<br />

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bvsms.saude.gov.br/bvs/saudelegis/gm/2012/<br />

prt0793_24_04_2012.html<br />

7. BRASIL, Presidência da República. Lei n° 12.303 de 02<br />

de agosto de 2010. Dispõe sobre a obrigatoriedade de<br />

realização do exame denominado Emissões Otoacústicas<br />

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8. Shargorodsky J, Curhan SG, Curhan GC, Eavey R. Change<br />

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9. BRASIL, Ministério da Saúde. 2009. Saúde na Escola - Série<br />

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basica_24.pdf<br />

10. McPherson B, Law MMS, Wong MSM. Hearing screening<br />

for school children: comparison of low-cost, computer-based<br />

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11. Yueh B, Shapiro N, MacLean CH, Shekelle PG. Screening<br />

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tele-audiometry. Telemed J E Health. 2007;13(5):501-8.<br />

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Um Método de Baixo Custo para Triagem Auditiva. Arq. Int.<br />

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D, Elangovan S. Internet-based tele-audiometry system for<br />

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hearing assessment - a study in tele-audiology. J. Telemed.<br />

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16. Food and Drug Administration (FDA). 1997. Code of<br />

Federal Regulations Title 21: Food and drugs chapter I -<br />

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cdrh/cfdocs/cfcfr/cfrsearch.cfm?cfrpart=11<br />

<strong>17</strong>. American Speech-Language-Hearing Association<br />

(ASHA). 1997. Guidelines for Audiologic Screening<br />

[Guidelines - Internet]. [cited 2012 Oct 12]. Available from.<br />

Available from: www.asha.<strong>org</strong>/policy.<br />

18. ANSI. Maximum Permissible Ambient Noise Levels for<br />

Audiometric Test Rooms. American National Standards<br />

Institute, S3.1-1999, 1999, New York.<br />

19. Landis, JR.; Koch, GG. The measurement of observer<br />

agreement for categorical data. Biometrics. 1977;33:159-74.<br />

20. Wang YF, Wang SS, Tai CC, Lin LC, Shiao AS. Hearing<br />

screening with portable screening pure-tone audiometer<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):265-273.<br />

DOI: 10.7162/S1809-97772013000300006<br />

Comparison of videonasoendoscopy and auditory-perceptual evaluation<br />

of speech in individuals with cleft lip/palate<br />

Lauren Medeiros Paniagua 1 , Alana Verza Signorini 2 , Sady Selaimen da Costa 3 , Marcus Vinicius Martins Collares 4 ,<br />

Sílvia Dornelles 5 .<br />

1) Speech Language Pathologist. Doctor of Science in Children’s and Teenager’s Health- Universidade Federal do Rio Grande do Sul-UFRGS. Professor of Speech-Language<br />

Pathology at Fatima College (RS).<br />

2) Undergraduated Speech pathology - Universidade Federal do Rio Grande do Sul - UFRGS (fellow undergraduated of CNPq).<br />

3) MD; MSc; PhD.Associate Professor - Department Of Otolaryngology & Head And Neck Surgery School Of Medicine - Universidade Federal do Rio Grande do Sul.<br />

4) MD; PhD. Associate Professor Department of Surgery; Head, Plastic Surgery Section - School of Medicine/Universidade Federal do Rio Grande do Sul.<br />

5) Speech Pathology, MSc, PhD. Professor Department of Speech Pathology - Universidade Federal do Rio Grande do Sul.<br />

Institution: Universidade Federal do Rio Grande do Sul (UFRGS).<br />

Porto Alegre / RS - Brazil.<br />

Mailing address: Lauren Medeiros Paniagua - Universidade Federal do Rio Grande do Sul (UFRGS) - Avenida João Wallig <strong>17</strong>05/627 – Porto Alegre / RS - Brazil - Zip Code:<br />

91340-001 - E-mail: lmedeirospaniagua@yahoo.com.br<br />

Article received on December <strong>17</strong> th , 2012. Article accepted on April 7 th , 2013.<br />

SUMMARY<br />

Introduction: The velopharyngeal sphincter (VPS) is a muscle belt located between the oropharynx and the nasopharynx.<br />

Investigations of velopharyngeal function should include an auditory-perceptual evaluation and at least 1 instrument-based<br />

evaluation such as videonasoendoscopy.<br />

Aim: To compare the findings of auditory-perceptual evaluation (hypernasality) and videonasoendoscopy (gap size) in individuals<br />

with cleft lip/palate.<br />

Method: This was a retrospective, cross-sectional study assessing 49 subjects, of both sexes, with cleft lip/palate followed up<br />

at the Otorhinolaryngology Service and the Speech Therapy outpatient clinic of Hospital de Clínicas de Porto Alegre (HCPA).<br />

The results from the auditory-perceptual evaluation and the videonasoendoscopy test were compared with respect to the VPS<br />

gap size.<br />

Results: Subjects with moderate/severe hypernasality had more severe velopharyngeal closure impairment than those with a<br />

less severe condition. The interaction between hypernasality severity and the presence of other speech disorders (p = 0.035),<br />

whether compensatory and/or obligatory, increased the likelihood of having a moderate-to-large gap in the velopharyngeal<br />

closure.<br />

Conclusions: We observed an association between the findings of these 2 evaluation methods.<br />

Keywords: Cleft Palate; Velopharyngeal Sphincter; Communication Disorders; Evaluation.<br />

INTRODUCTION<br />

The velopharyngeal sphincter (VPS) is a muscle belt<br />

located between the oropharynx and the nasopharynx and<br />

includes the muscles of the soft palate (anterior wall of the<br />

VPS) and the lateral and posterior pharyngeal walls. The<br />

VPS comprises the levator veli palatini, tensor veli palatini,<br />

muscle of uvula, superior pharyngeal constrictor,<br />

palatopharingeus, palatoglossus, and salpingopharyngeus<br />

(1). Among these muscles, those of the soft palate,<br />

especially the tensor veli palatine, have an important role<br />

in the physiological function of the region. While the VPS<br />

is not a sphincter by strict definition (a circular muscle band<br />

in a hollow viscera), its physiological function is that of a<br />

sphincter, as it works like a valve, contracting and occluding<br />

itself (2).<br />

The VPS plays an extremely important role in<br />

allowing alternation between the respiratory and digestive<br />

paths of the pharynx during swallowing (3). Velopharyngeal<br />

closure, performed by the VPS, is essential in motor actions<br />

of the region such as speaking, whistling, blowing, sucking,<br />

and swallowing (4). Several studies have described intersubject<br />

variability in velopharyngeal closure (1, 5) and<br />

have identified 4 types of closure based on structure<br />

movement (6, 7, 1, 5). In the coronal type of closure, there<br />

is more participation of the soft palate. Sagittal closure<br />

predominantly involves the lateral pharyngeal walls. In the<br />

circular type of closure, the soft palate and lateral pharyngeal<br />

walls are involved. Finally, the circular type closure with a<br />

Passavant ridge, is similar to the circular closure except that<br />

a Passavant ridge is present on the posterior pharyngeal<br />

wall.<br />

The configuration of the velopharyngeal region<br />

depends on the specific motor actions performed. The<br />

raising of the soft palate and, concomitantly, the medial<br />

approximation of the lateral pharyngeal walls promotes<br />

the partial or complete separation of the nasal and oral<br />

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Paniagua et al.<br />

portions of the pharynx. The velopharyngeal structures are<br />

thus important for the production of speech, as they<br />

distribute and direct airflow to the oral cavity to produce<br />

oral sounds and to the nasal cavity for the emission of nasal<br />

sounds (1, 8, 9).<br />

Investigators have several types of tools to choose<br />

from when assessing VPS function (10, 11, 12). A detailed<br />

description of the velopharyngeal mechanism was obtained<br />

only after the availability of both direct and indirect<br />

assessment methods. Direct methods enable the investigator<br />

to visualize the structures involved in velopharyngeal<br />

closure and to observe how these structures move during<br />

different types of motor actions. Indirect methods provide<br />

information on the functional repercussions of<br />

velopharyngeal action (13, 12, 14).<br />

Videonasoendoscopy is one of the most widely<br />

used methods for studying velopharyngeal function,<br />

allowing visualization of the nasal, pharyngeal, and laryngeal<br />

cavities. Using dynamic images directly from the anatomical<br />

structures (15), this method has multiple applications,<br />

including diagnosis, prognosis, and postoperative follow<br />

up (11). This test can be used to observe the pattern of<br />

closure even during speech, which involves wide variations<br />

in velum and pharyngeal wall motion. Videonasoendoscopy<br />

can also be used to identify the presence of the gap<br />

corresponding to the residual orifice during maximum<br />

contraction of the VPS (16, <strong>17</strong>).<br />

As a counterpart to the use of more sophisticated<br />

tools for velopharyngeal assessment, clinical judgment by<br />

means of hearing is still considered an extremely important<br />

diagnostic tool for inferring velopharyngeal function (12).<br />

Using this method, the investigator can assess a subject’s<br />

oral communication performance and determine the extent<br />

of speech impairments related to resonance (18, 12, 19,<br />

20, 21).<br />

Impaired velopharyngeal closure prevents separation<br />

of the oral and nasal cavities during the production of oral<br />

phonemes, exposing the nasal cavity to the entry of<br />

unexpected air flow. This impairment in velopharyngeal<br />

closure, known as velopharyngeal dysfunction (VPD), can<br />

be congenital, as with cleft lip.<br />

There is a widespread consensus among researchers<br />

and healthcare professionals that the surgical and clinical<br />

findings of subjects with cleft lip/palate should be based<br />

on an auditory-perceptual evaluation and at least one<br />

instrument-based evaluation of velopharyngeal function<br />

(13, 22, 23, 24). The use of both assessment modalities<br />

is highly valuable and widely used in clinical practice, as<br />

the findings of one method complement the other.<br />

Despite this common practice, there are few studies<br />

comparing patient evaluations using videonasoendoscopy<br />

(clinical estimation of gap size) and auditory-perceptual<br />

assessments (hypernasality). This study aimed to determine<br />

whether the findings of videonasoendoscopy in<br />

patients with cleft lip/palate are associated with<br />

demographic features, clinical factors, or the findings of<br />

auditory-perceptual assessments.<br />

METHODS<br />

This retrospective, cross-sectional study assessed<br />

49 individuals, of both genders, with cleft lip/palate who<br />

were followed up at the Otorhinolaryngology Service and<br />

at the Speech Therapy outpatient clinic of Hospital de<br />

Clínicas de Porto Alegre (HCPA). The study was approved<br />

by the HCPA Research Ethics Committee under protocol<br />

number 10-0490.<br />

The population consisted of individuals follow up at<br />

the HCPA in the Plastic Surgery Service, Craniofacial<br />

Surgery outpatient clinic; Otorhinolaryngology and at the<br />

“SpeechTherapy and Craniofacial Malformations” outpatient<br />

clinic. This group of patients was primarily treated at HCPA<br />

and at other institutions.<br />

The present study compared the results of 2 methods<br />

for evaluating velopharyngeal function. The auditoryperceptual<br />

evaluation by speech therapy screening is<br />

reported as speech findings. This assessment was carried<br />

out by a speech therapist with more than 10 years of<br />

experience in the area. The results of videonasoendoscopy<br />

were analyzed by 2 experienced evaluators. The speech<br />

therapy screening and videonasoendoscopy data were<br />

collected in October 2012. The inclusion criterion was<br />

assessment by speech therapy screening and<br />

videonasoendoscopy between September 2011 and August<br />

2012. We excluded from the study patients with blurred<br />

videonasoendoscopy images and those with excess<br />

secretion from the nasal cavity or missing data.<br />

The speech therapy screening assessed the following<br />

auditory-perceptual parameters: resonance, presence, and<br />

severity of hyponasality and hypernasality (mild, moderate,<br />

severe) (25); speech disorders, identified as compensatory<br />

(such as glottal stops) or obligatory (weak intraoral pressure,<br />

facial mimicry, audible nasal air emission, nasal snoring).<br />

For analysis purposes, hypernasality and its severity were<br />

included as obligatory disorders (26). During screening, the<br />

speech therapist used a speech sample comprising 2<br />

sentences with phonetic predominance of oral plosive<br />

phonemes/p/ (“papai pediu pipoca”) and fricative<br />

phonemes/s/ (“o saci sabe assobiar” The patients also<br />

performed a segment of continuous speech, counting from<br />

1 to 10.<br />

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Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with cleft lip/palate.<br />

Paniagua et al.<br />

Using videonasoendoscopy, we analyzed the portion<br />

of the test related to the sustained emission of the /s/<br />

phoneme, chosen because it leads to complete closure of<br />

the VPS, thereby minimizing misinterpretations due to<br />

interference by the production of another phoneme. The<br />

evaluators made a clinical estimation of the VPS gap size<br />

using an adapted version of a protocol described in previous<br />

studies (10, 27). The VPS gap size was classified according<br />

to the following scale: 1, no gap (complete velopharyngeal<br />

closure); 0.8–0.9, small gap (efficient closure); 0.4–0.7,<br />

moderate gap (intermediate closure); 0.1–0.3 large gap<br />

(inefficient closure); 0, very large gap (lack of closure). This<br />

protocol assessed the following: presence or absence of<br />

adenoids contributing to velopharyngeal closure; presence<br />

or absence of mobility of the palate and lateral and posterior<br />

pharyngeal walls; VFS closure type (coronal, sagittal, circular,<br />

or circular with Passavant ridge).<br />

Quantitative variables were described as mean and<br />

standard deviation or median and interquartile range.<br />

Categorical variables were described as absolute or relative<br />

frequencies. The Pearson’s chi-square test was used to<br />

evaluate the association between videonasoendoscopy<br />

and perceptual-hearing evaluation. The McNemar’s test<br />

was applied to assess the differences between evaluators,<br />

and the kappa coefficient was applied to assess interevaluator<br />

agreement. Student’s t-test (quantitative variables<br />

with symmetric distribution), Mann–Whitney test<br />

(quantitative variables with asymmetric distribution), and<br />

Pearson’s chi-square and Fisher’s exact tests (categorical<br />

variables) were applied to investigate the association<br />

between clinical factors and examination methods. Poisson’s<br />

regression model was used to control confounding factors.<br />

The criterion for including a variable in the model was a p<br />

value < 0.10 in the bivariate analysis. We considered<br />

significance to be 5% (p d” 0.05). Analyses were performed<br />

using SPSS (Statistical Package for the Social Sciences)<br />

version <strong>17</strong>.0.<br />

RESULTS<br />

The median age at the time of evaluation was 11<br />

years, with the majority of the cohort between 9 and 16<br />

years. The cohort was predominantly male (61.2%). Speech<br />

therapy treatment was ongoing in 14.3% of the subjects.<br />

The most common cleft type was the left unilateral<br />

complete cleft (30.6%, n = 15), followed by bilateral<br />

complete cleft lip/palate (28.6%, n = 14), right unilateral<br />

(20.4%, n = 10), cleft palate (14.3%, n = 7), and submucosal<br />

cleft (6.1 %, n = 3).<br />

All patients had undergone primary palatoplasty at<br />

a mean age of 19.5 months. Secondary palatoplasty was<br />

performed in only 4.1% of the subjects, at a median age of<br />

18.5 years.<br />

Table 1 shows the results from the speech therapy<br />

screening, including the assessment of resonance as well as<br />

compensatory and obligatory articulation disorders. We<br />

found that 77.6% of the subjects presented with<br />

hypernasality, which was moderately severe in 36.7% and<br />

mildly severe in 28.6% of these subjects. As illustrated in<br />

Table 1, articulation disorders were found in 81.6% of<br />

subjects, 75.5% of whom had obligatory disorders and<br />

57.1% of whom had compensatory disorder (glottal stops).<br />

The most frequently observed obligatory disorder was<br />

weak intraoral pressure, which was present in 75.7% of the<br />

subjects. The second more frequent disorder was audible<br />

nasal air emission (62.2%).<br />

Inter-evaluator comparison of the findings of the<br />

videonasoendoscopy, as well as comparison of these test<br />

results with those of the auditory-perceptual evaluation<br />

was assessed using the emission of the /s/ phoneme.<br />

The inter-rater agreement was established using the<br />

Kappa agreement coefficient, matching the findings of<br />

one rater with those of the other. In this study, the Fleiss’<br />

kappa agreement coefficients (28) were interpreted as<br />

previously reported (29):


Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with cleft lip/palate.<br />

Paniagua et al.<br />

velopharyngeal closure, mobility of pharyngeal walls,<br />

predominant type of movement, closure type, and VPS<br />

gap size during emission of the /s/ phoneme. The<br />

predominant closure type was coronal. One evaluator’s<br />

results indicated that 20.4% of the subjects had no VPS gap,<br />

40.8% had a small gap, 16.3% had a moderate gap, and<br />

22.4% had a large gap.<br />

Demographic data indicated that the subjects with<br />

more severe velopharyngeal closure impairment underwent<br />

later primary palatoplasty; the presence of a moderate to<br />

large gap was significantly associated with an age at<br />

primary palatoplasty of over 23 months. All individuals<br />

requiring secondary palatoplasty (n = 2) showed impairment<br />

in velopharyngeal closure, although this result was not<br />

statistically significant. There were no other statistically<br />

significant associations between variables.<br />

We compared the VPS gap size as estimated by<br />

clinical and auditory-perceptual assessments (Table 2). We<br />

also investigated the relationship between the gap size and<br />

both the severity of hypernasality and the presence of<br />

speech disorders (compensatory and obligatory disorders).<br />

Our results indicate that more severe impairment of<br />

velopharyngeal closure was observed more often in subjects<br />

with moderate/severe hypernasality, with statistical<br />

significance for severe hypernasality (p = 0.021). In addition,<br />

we found severe hypernasality was significantly associated<br />

with the presence of other speech disorders (p = 0.035),<br />

whether compensatory and/or obligatory, increasing the<br />

likelihood of having a moderate to large gap (intermediate<br />

to inefficient velopharyngeal closure).<br />

Ten subjects (20.4%) showed discrepant results<br />

with regard to velopharyngeal function, having moderate/<br />

severe hypernasality + other speech disorders together<br />

with complete and efficient gap closure. Conversely, 6<br />

subjects (12.2%) had intermediate/inefficient closure with<br />

no moderate/severe hypernasality.<br />

These discrepant findings may be due to the<br />

differences in the analytical parameters (speech and<br />

continuous sound emission), although both parameters<br />

involve velopharyngeal function. The auditory-perceptual<br />

evaluation assessed a larger speech sample (counting of<br />

numbers and 2 sentences focusing on /s/ and /p/<br />

phonemes). The instrumental evaluation observed the<br />

sustained emission of the /s/ phoneme, which provides<br />

maximum VPS contraction. In such cases that present<br />

physical evidence of closure but indicate abnormal<br />

resonance, it is very important for the interdisciplinary<br />

team to re-evaluate the findings to perform a differential<br />

diagnosis of velopharyngeal dysfunction. Therefore, it can<br />

be inferred that the maintenance of motor action in speech<br />

is not consistent.<br />

Table 2. Relationship between auditory-perceptual assessment and gap size.<br />

Variables Complete/efficient closure Intermediate/inefficient closure p<br />

Speech disorder, n (%) 0.636<br />

Present 26 (86.7) 18 (94.7)<br />

Absent 4 (13.3) 1 (5.3)<br />

Moderate/severe hypernasality +<br />

other speech disorders, n (%) 0.035<br />

Yes 10 (33.3) 13 (68.4)<br />

No 20 (66.7) 6 (31.6)<br />

Type of disorder, n (%) 0.157<br />

Obligatory 12 (46.2) 4 (22.2)<br />

Compensatory 1 (3.8) 0 (0.0)<br />

Obligatory + compensatory 13 (50.0) 14 (77.8)<br />

Types of obligatory disorders*, n (%)<br />

Audible nasal air emission 13 (43.3) 10 (52.6) 0.733<br />

Weak intraoral pressure <strong>17</strong> (56.7) 11 (57.9) 1.000<br />

Facial mimicry 4 (13.3) 7 (36.8) 0.081<br />

Nasal snoring 2 (6.7) 0 (0.0) 0.515<br />

Hypernasality 21 (70.0) <strong>17</strong> (89.5) 0.165<br />

Resonance, n (%) 0.021<br />

Balanced 9 (30) 2 (10.5)<br />

Mild hypernasality 11 (36.7) 3 (15.8)<br />

Moderate hypernasality 9 (30) 9 (47.4)<br />

Severe hypernasality 1 (3.3) 5 (26.3)**<br />

*multiple choice question; **statistically significant association by the test of residuals adjusted to a 5% of significance level.<br />

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Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with cleft lip/palate.<br />

Paniagua et al.<br />

DISCUSSION<br />

The physiology of the velopharyngeal mechanism<br />

has been investigated in several disciplines (30, 31).<br />

Studies comparing different methods of assessing<br />

velopharyngeal function report conflicting results. Few of<br />

these investigations have considered the following factors<br />

in the same study: (1) protocols for clinical and instrumentbased<br />

assessments recommended in the literature, including<br />

a detailed description of the criteria used for analysis of the<br />

variables under study; (2) inter-evaluator agreement for<br />

assessments using videonasoendoscopy; (3) the possible<br />

variables interfering with velopharyngeal closure<br />

(participation of adenoids and speech therapy treatment<br />

for velopharyngeal occlusion).<br />

Our study assessed hypernasality, compensatory<br />

articulation disorders, and other obligatory disorders (26).<br />

These speech anomalies are of interest to researchers who<br />

investigate speech production in individuals with cleft lip/<br />

palate (32, 33 34, 35, 25).<br />

A recently study (26) has emphasized that obligatory<br />

disorders should receive attention from a structural point of<br />

view and not only from the perspective of velopharyngeal<br />

function; the identification of velopharyngeal dysfunction<br />

can help to establish whether speech therapy intervention<br />

is indicated for individuals with cleft lip/palate. The auditoryperceptual<br />

evaluation used in the present study took these<br />

structural features of obligatory disorders into account.<br />

Hypernasality was reported in 77.6% of our subjects, the<br />

severity of which was moderate in 36.7% and mild in<br />

28.6%. Speech disorders were identified in 89.8% of the<br />

subjects, of whom 87.8% had obligatory disorders and<br />

57.1% compensatory disorder (glottis stop). One of the<br />

most frequent obligatory disorders was weak intraoral<br />

pressure, present in 75.7% of the subjects. Our findings<br />

corroborate those in the literature reporting a higher<br />

occurrence of weak intraoral pressure, hypernasality, and<br />

glottis stop (37, 4, 37). The results of this study thus suggest<br />

that the weak intra-oral pressure caused by VPD produces<br />

audible nasal air emission and hypernasality in speech. In<br />

turn, glottis stops may occur as a consequence of poor<br />

articulation habits learned in childhood that do not necessarily<br />

reflect physical or neuromuscular changes (38).<br />

We believe that the high occurrence of hypernasality<br />

and other articulation disorders in our cohort is related to their<br />

referrals from speech therapy, indicating that they had an<br />

issue involving oral communication. Although these patients<br />

were followed up for a longer time by the HCPA<br />

interdisciplinary team that provides a variety of services, a<br />

speech therapy outpatient clinic linked to Universidade<br />

Federal do Rio Grande do Sul was added in 2011 to treat<br />

patients with cleft lip/palate. Before the creation of this<br />

outpatient clinic, the patients were treated just by a speech<br />

therapist from HCPA, with a period of care limited to one<br />

shift for this population. As it constitutes a curricular internship,<br />

in which undergraduate and graduate students practice the<br />

activity, the response to this demand is still limited.<br />

Reports in the literature point out that experience<br />

is crucial in both the conduction and analysis of the<br />

videonasoendoscopy test. Accordingly, we assessed the<br />

inter-evaluator agreement with regard to gap size in the<br />

emission of the /s/ phoneme, observing that it was almost<br />

perfect (kappa, 0.83–0.95). We thus conclude that the<br />

evaluation team is highly qualified. This result also illustrates<br />

that the criteria adopted for interpreting the findings were<br />

very homogeneous between the raters. These findings<br />

corroborate those described in other scientific studies using<br />

this protocol (10), indicating that the data are in accordance<br />

with the proposed objective (39, 40, 41).<br />

We observed that 20.4% of the subjects did not have<br />

a VPS gap; 40.8% had a small gap, 16.3% had a moderate<br />

gap, and 22.4% had a large gap. Thus, 61.2% of the total<br />

cohort had complete or efficient velopharyngeal closure.<br />

The predominant velopharyngeal closure type was coronal<br />

(69.4%, n = 34) which has greater mobility of the anterior<br />

wall of the VPS corresponding to the soft palate. The<br />

prevalence of the sagittal type was 24.5% (12), and the<br />

circular type was 6.1% (3). Studies have demonstrated that<br />

the coronal pattern is also the most frequent among individuals<br />

without cleft lip/palate (42, 22, 30, 7), but individuals with<br />

cleft palate tend to have a broader distribution between the<br />

other patterns of closure (43, 7, 44).<br />

Among the associations related to demographic and<br />

clinical data, only age at the time of primary palatoplasty<br />

was associated with the clinical estimation of VPS gap size.<br />

Our findings indicate that the later primary palatoplasty is<br />

performed the greater the compromise in velopharyngeal<br />

closure (moderate to large gap size), with a significant<br />

associated with ages above 23 months (p = 0.016).<br />

However, when analyzing the variables independently<br />

associated with intermediate/inefficient velopharyngeal<br />

closure (by controlling confounding factors), age at primary<br />

palatoplasty was not significantly associated but was near<br />

the threshold (PR = 1.01; 95% CI, 1.00–1.02; p = 0.088).<br />

The trend in the data indicates that every additional month<br />

until the performance of the first palatoplasty leads to a 2%<br />

increase in the prevalence of inefficient closure.<br />

The author of a previous study (46) proposes that<br />

the appropriate time for surgical repair of the palate is 12–<br />

18 months of age, taking into account maxillary growth and<br />

velopharyngeal function. One report on the association<br />

between various clinical factors and velopharyngeal closure<br />

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Comparison of videonasoendoscopy and auditory-perceptual evaluation of speech in individuals with cleft lip/palate.<br />

Paniagua et al.<br />

in cleft lip/palate found that velopharyngeal closure was<br />

higher among individuals with deciduous dentition than<br />

among those with mixed or permanent dentition (47).<br />

The present study aimed to compare the findings of<br />

auditory-perceptual evaluation and those of<br />

videonasoendoscopy. Our data indicate that subjects with<br />

more compromised velopharyngeal closure have a higher<br />

incidence of moderate and severe hypernasality, showing<br />

that both tests indicated the compromise in velopharyngeal<br />

function. A statistically significant association was found<br />

between severe hypernasality (p = 0.021) and intermediate/<br />

inefficient closure with a gap size ranging from moderate<br />

to large.<br />

We found one study that aimed to evaluate auditoryperceptual<br />

assessment, but the authors adopted a different<br />

classification system for the findings of videonasoendoscopy<br />

and did not relate them to the efficiency of velopharyngeal<br />

closure. A retrospective study investigated the relationship<br />

between the auditory-perceptual characteristics of speech<br />

and velopharyngeal gap size (20), and the authors concluded<br />

that information regarding velopharyngeal gap size can be<br />

predicted from speech assessment alone. Confidence in<br />

the prediction was stronger when the patient had moderate<br />

to severe hypernasality, which was more commonly<br />

associated with a large gap. This last finding was similar to<br />

that reported in our study.<br />

We found a statistically significant interaction (p =<br />

0.035) of the severity of hypernasality and the presence of<br />

other articulation disorders with the clinical estimate of VPS<br />

gap size. The interaction of both changes identified in the<br />

auditory-perceptual evaluation increased the likelihood of<br />

the presence of a moderate to large gap. Multivariate<br />

analysis confirmed this association (PR = 1.26; 95% CI:<br />

1.03–1.54; p = 0.026). This result is observed in the clinical<br />

practice, as the presence of hypernasality is usually<br />

associated with at least one articulation change, whether<br />

obligatory or compensatory. The articulation changes<br />

associated with hypernasality reflect the difficulty in<br />

acquiring and maintaining velopharyngeal closure (47).<br />

To gain a better understanding of these cases, we<br />

investigated velopharyngeal physiology during the motor<br />

action related to speech. Although no statistically significant<br />

difference was found, 2 interesting situations were observed.<br />

First, 10 subjects (20.4%) had complete/efficient<br />

velopharyngeal closure but also had moderate/severe<br />

hypernasality. Second, 6 subjects (12.2%) had intermediate/<br />

inefficient closure with no moderate/severe hypernasality.<br />

Although both videonasoendoscopy and auditoryperceptual<br />

evaluation assess velopharyngeal function, this<br />

study used different samples and speech extensions. The<br />

videonasoendoscopy analyzed only the sustained emission<br />

of the /s/ phoneme, using a comfortable intensity for the<br />

patient that was audible for the examiner. This phoneme<br />

was chosen because its emission requires the action of all<br />

velopharyngeal walls and their maximum contraction to<br />

achieve complete velopharyngeal closure. This information<br />

allows the examiner to identify the presence or absence of<br />

a VPS gap.<br />

Velopharyngeal closure differs during different motor<br />

actions and during speech (4). The observation that some<br />

patients have unsatisfactory findings in the auditoryperceptual<br />

evaluation and good findings in<br />

videonasoendoscopy suggests that resonance in continuous<br />

speech is unaffected. However, articulation disorders can<br />

hamper accurate assessment of hypernasality severity.<br />

Hypernasality is often associated with the length of time<br />

required for VPS opening and not directly with the degree<br />

of opening or even with the air flow that escapes from the<br />

nasal cavity (48). For example, patients with adequate<br />

velopharyngeal closure as assessed by instrument-based<br />

assessment may have hypernasality because of the<br />

abnormalities in the temporal spectrum of velopharyngeal<br />

closure.<br />

The great physiological variability of the VPS may<br />

be related to factors such as speed of speech, characteristics<br />

of isolate phoneme emission, and association of different<br />

phonemes (consonant-vowel) (1). This variability may<br />

account for the patients who have intermediate/inefficient<br />

closure but no moderate/severe hypernasality. Further<br />

factors may explain this versatility in velopharyngeal<br />

closure as follows: the action of several muscles associated<br />

with the levator veli palatini (palatoglossus,<br />

palatopharingeus, and superior pharyngeal constrictor);<br />

mechanical factors, especially the position of the tongue in<br />

the oral cavity; and the specific phonological rules of each<br />

linguistic system (49, 5).<br />

In summary, we recommend that patients with<br />

discrepant results between evaluations and those with<br />

unsatisfactory results should be referred to interdisciplinary<br />

clinical evaluations to determine the best course of action.<br />

(26). X et al. Emphasizes that obligatory and compensatory<br />

articulation disorders affect speech, and that obligatory<br />

disorders should receive attention from both the structural<br />

and functional points of view (26). Therefore, by identifying<br />

velopharyngeal dysfunction, appropriate therapeutic<br />

strategies can be devised.<br />

CONCLUSIONS<br />

It was found that our patient cohort had a high<br />

prevalence of hypernasality and compensatory and<br />

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Paniagua et al.<br />

articulation disorders. In addition, we observed a high<br />

frequency of individuals with a small gap, and a<br />

predominance of the coronal type closure. The individuals<br />

with more severe velopharyngeal closure impairment<br />

underwent late palatoplasty, although this comparison did<br />

not remain significant after adjustment by the multivariate<br />

model.<br />

Comparison of the 2 evaluation methods showed<br />

that subjects with moderate/severe hypernasality had<br />

more severely impaired velopharyngeal closure.<br />

Hypernasality is more closely associated with clinical<br />

estimation of gap size than are other obligatory and<br />

compensatory disorders. The interaction between<br />

hypernasality severity and the presence of other disorders<br />

increases the likelihood of having a gap ranging from<br />

moderate to large. Although we found cases with<br />

discrepancies between the findings of the auditoryperceptual<br />

and instrument-based evaluations, we observed<br />

an association between the findings of these 2 evaluation<br />

methods.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):274-278.<br />

DOI: 10.7162/S1809-97772013000300007<br />

Clinical and fiberoptic endoscopic assessment of swallowing in patients<br />

with chronic obstructive pulmonary disease<br />

Marina Rodrigues Bueno Macri 1 , Jair Mendes Marques 2 , Rosane Sampaio Santos 3 , Ana Maria Furkim 4 , Irinei Melek 5 ,<br />

Daniel Rispoli 6 , Maria Cristina de Alencar Nunes 7 .<br />

1) Speech Therapist. Masters in Communication Disorders from the University of Paraná Tuiuti.<br />

2) Degree in Mathematics. Doctorate in Geodetic Sciences from the Federal University of Paraná (HC-UFPR).<br />

3) Speech Therapist. Student Doctorate in Internal Medicine from the HC-UFPR.<br />

4) Speech Therapist. Doctorate in Human Communication Disorders from the Federal University of Sao Paulo.<br />

5) Doctor. Pulmonologist.<br />

6) Doctor. Otolaryngologist.<br />

7) Speech Therapist. Student Doctorate in Internal Medicine from the HC-UFPR.<br />

Institution: University of Paraná Tuiuti.<br />

Curitiba / PR - Brazil.<br />

Mailing address: Marina Rodrigues Bueno Macri - Rua Saldanha da Gama 86, apt. 42b - Curitiba / PR - Brazil - Zip code: 80060-<strong>17</strong>0 - E-mail: buenomarina405@hotmail.com<br />

Article received on January <strong>17</strong> th , 2013. Article accepted on April 7 th , 2013.<br />

SUMMARY<br />

Introduction: Chronic obstructive pulmonary disease is characterized by progressive and partially reversible obstruction of<br />

pulmonary airflow.<br />

Aim: To characterize swallowing in patients with chronic obstructive pulmonary disease and correlate the findings with the<br />

degree chronic obstructive pulmonary disease, heart and respiratory rate, oxygen saturation, and smoking.<br />

Method: We conducted a prospective cohort study of 19 patients (12 men and 7 women; age range, 50–85 years) with confirmed<br />

medical diagnosis of chronic obstructive pulmonary disease. This study was performed in 2 stages (clinical evaluation and functional<br />

assessment using nasolaryngofibroscopy) on the same day. During both stages, vital signs were checked by medical personnel.<br />

Results: Clinical evaluation of swallowing in all patients showed the clinical signs of cough. The findings of nasolaryngofibroscopy<br />

highlighted subsequent intraoral escape in 5 patients (26.5%). No patient had tracheal aspiration. There was no association of<br />

subsequent intraoral escape with degree of chronic obstructive pulmonary disease, heart and respiratory rate, oxygen saturation,<br />

or smoking.<br />

Conclusion: In patients with chronic obstructive pulmonary disease, there was a prevalence of oral dysphagia upon swallowing<br />

and nasolaryngofibroscopy highlighted the finding of subsequent intraoral escape. There was no correlation between intraoral<br />

escape and the degree of chronic obstructive pulmonary disease, heart and respiratory rate, oxygen saturation, or smoking.<br />

Keywords: Pulmonary Disease, Chronic Obstructive; Deglutition Disorders; Respiration; Smoking; Heart Rate; Respiratory Rate.<br />

INTRODUCTION<br />

Chronic obstructive pulmonary disease (COPD) is<br />

characterized by progressive and partially reversible airway<br />

obstruction. This airflow obstruction is associated with an<br />

abnormal inflammatory response of the lungs and occurs<br />

between 5% and 15% of the adult population (1-3) and is<br />

a leading cause of death worldwide. Smoking is responsible<br />

for more than 90% of the cases, with a prevalence of<br />

disease found in men (4).<br />

The diagnosis of COPD should be considered in the<br />

presence of cough, sputum production, dyspnea, and a<br />

history of exposure to risk factors such as smoking,<br />

environmental pollution, and occupational exposure to<br />

toxic gases or particles (1).<br />

Several factors can lead to inadequate food intake in<br />

patients with COPD, causing weight loss, oropharyngeal<br />

dysphagia (possibly due to difficulty in chewing), dyspnea,<br />

cough, fatigue, and secretion (5).<br />

Oropharyngeal dysphagia is a swallowing disorder<br />

with specific signs and symptoms that are characterized by<br />

alterations at any stage and/or between the steps of the<br />

dynamics of swallowing; this condition may be congenital<br />

or acquired (6,7). Stable dysphagic patients with COPD can<br />

present important clinical complications such as malnutrition,<br />

pulmonary complications, dehydration, and discomfort<br />

when eating (8,9).<br />

In patients with oropharyngeal dysphagia, clinical<br />

evaluation of swallowing associated with instrumental<br />

evaluation techniques such as nasolaryngofibroscopy (FEES)<br />

(10) is required, which assists in the application of therapeutic<br />

approaches.<br />

The present study aimed to characterize swallowing<br />

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Macri et al.<br />

in patients with COPD and correlate the findings of<br />

swallowing with the degree of COPD, heart and respiratory<br />

rate, oxygen saturation, and smoking.<br />

METHOD<br />

A prospective cohort study was performed in 19<br />

patients with confirmed diagnosis of COPD. Seven patients<br />

(40.0%) were female and 12 patients (60.0%) were male.<br />

Patient age ranged between 50 and 85 years, with a mean<br />

of 66.7 years. This study was approved by the research<br />

ethics committee of our institution (No. OF. CEP Hospital<br />

Angelina Caron 42/09). Subjects signed a consent form<br />

after being informed of the objectives, procedures, and<br />

responsibilities of the study, as well as receiving answers to<br />

any questions regarding the study.<br />

Inclusion criteria included patients of both genders<br />

aged over 50 years with a diagnosis of COPD according to<br />

the GOLD scale (11), who had not used inhaled medication<br />

up to 4 h before evaluations, and were smokers and nonsmokers.<br />

Patients with a lowered level of consciousness<br />

and clinically unstable patients were excluded from the<br />

study.<br />

The time for COPD diagnosis was 5.7 years on<br />

average, with a minimum of 2 months, a maximum of 12<br />

years, and a standard deviation of 10.0 years. Of the 19<br />

patients, 14 (75.0%) were smokers with a mean duration<br />

of smoking of 41.1 years and a standard deviation of<br />

smoking of 5.0 years. The remaining 5 patients (25.0%)<br />

had never smoked.<br />

The study was conducted in 2 stages on the same<br />

day. Patients referred to the pulmonology clinic with a<br />

clinical diagnosis of COPD were transferred to the<br />

otolaryngology clinic of the same hospital, where the tests<br />

were conducted. The first stage consisted of clinical<br />

evaluation of swallowing and the second stage consisted of<br />

functional assessment of swallowing using FEES (10), with<br />

vital signs being checked by the medical team during both<br />

assessments.<br />

During the first stage, the protocol data for clinical<br />

evaluation of swallowing used in our hospital were followed.<br />

The clinical signs of aspiration (cough, dyspnea, and “wet”<br />

voice) were recorded (12). The food consistencies used<br />

(liquid, nectar, honey, pudding, and solid) followed the<br />

pattern of the American Dietetic Association (ADA) (13).<br />

For this evaluation, patients completed a sequence of 3<br />

swallows (free swallow, 5 mL, and 10 mL) for each food<br />

consistency. Swallowing function was evaluated by uptake<br />

of the bolus, closing of the lips, preparation of the bolus,<br />

subsequent extraoral exhaust, waste in the oral cavity,<br />

throat clearing, and coughing reflex. After evaluation, the<br />

Functional Oral Intake Scale (FOIS) (14) was applied to<br />

assess the level of food intake of each patient.<br />

During the second stage, FEES (10) was performed<br />

by otolaryngologists following the protocol data for FEES<br />

evaluation of swallowing used in our hospital. We used the<br />

same consistency of foods offered during the clinical<br />

evaluation of swallowing function, plus the in<strong>org</strong>anic dye<br />

aniline blue to contrast with the pink color of the mucosa.<br />

During examination, the presence of exhausted intraoral<br />

posterior pharyngeal residue in the posterior pharyngeal<br />

wall, epiglottic vallecula, and piriform sinus as well as<br />

pharyngeal clearance (number of swallows needed for<br />

clearance) and laryngeal penetration or tracheal aspiration<br />

(with or without reflex cough) were observed. The Severity<br />

Scale for Dysphagia: Penetration and Aspiration (15) was<br />

applied to the results.<br />

RESULTS<br />

Regarding respiratory impairment, 16 patients<br />

(85.0%) were in ambient air and 3 patients (15.0%) were<br />

oxygen dependent, one (5.0%) with 2 liters of oxygen,<br />

one (5.0%) with 3 liters of oxygen, and one (5.0%) with 5<br />

liters of oxygen. Regarding breathing pattern, 4 patients<br />

(20.0%) had wheezing, 1 (5.0%) showed mouth breathing,<br />

4 (20.0%) were mixed, 1 (5.0%) had tachypnea, and 9<br />

(50.0%) had dyspnea.<br />

Upon clinical evaluation of swallowing, all patients<br />

showed clinical signs of cough. Regarding food intake, all<br />

19 patients (100.0%) were at level 7 (oral full and<br />

unrestricted) of the FOIS. FEES findings indicated that<br />

exhausted intraoral posterior residue prevailed in 5 patients<br />

(26.5%) and no patient had tracheal aspiration. Residue in<br />

the epiglottic vallecula occurred in 1 patient (5.0%) with<br />

liquid and in 2 patients (10.5%) with solid food, both with<br />

bleaching.<br />

At the significance level of 0.05, there was no<br />

significant relationship between subsequent intraoral escape<br />

and the degree of COPD, heart and respiratory rate,<br />

oxygen saturation, or smoking, which can be observed in<br />

Tables 1, 2, 3, 4, and 5, respectively. The Severity Scale for<br />

Dysphagia: Penetration and Aspiration score was 1 (contrast<br />

does not enter the airway) in all 19 patients (100.0%).<br />

DISCUSSION<br />

Defining the incidence of oropharyngeal dysphagia<br />

in patients with COPD, as well as the identification of<br />

possible risk factors, may be useful in the management and<br />

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Macri et al.<br />

Table 1. Relationship between the degree of copd and intraoral escape.<br />

COPD GRADE<br />

INTRAORAL ESCAPE<br />

Pudding Liquid Solid<br />

A P A P A P<br />

II 2 (10.5%) - 1 (5.0%) 1 (5.0%) 2 (10.5%) -<br />

III 8 (42.5%) 1 (5.0%) 9 (47.5%) - 7 (37.0%) 2 (10.5%)<br />

IV 7 (37.0%) 1 (5.0%) 6 (31.5%) 2 (10.5%) 5 (26.0%) 3 (16.0%)<br />

p 0.6784 0.3756 0.3359<br />

COPD = chronic obstructive pulmonary disease, A = absent, P = present.<br />

Fisher’s test at a significance level of 0.05.<br />

Table 2. Relationship between heart rate and intraoral escape.<br />

HR<br />

INTRAORAL ESCAPE<br />

(bpm) Pudding Liquid Solid<br />

A P A P A P<br />

Less than 80 2 (10.5%) 1 (5.0%) 3 (16.0%) - 2 (10.5%) 1 (5.0%)<br />

80–89 7 (37.0%) - 7 (37.0%) - 6 (31.5%) 1 (5.0%)<br />

90–99 6 (31.5%) - 3 (16.0%) 3 (16.0%) 5 (26.5%) 1 (5.0%)<br />

100 or more 2 (10.5%) 1 (5.0%) 3 (16.0%) - 1 (5.0%) 2 (10.5%)<br />

p 0.7368 0.0867 0.4443<br />

HR = heart rate, bpm = beats per minute, A = absent, P = present.<br />

Fisher’s test at a significance level of 0.05.<br />

Table 3. Relationship between respiratory rate and intraoral escape.<br />

RR<br />

INTRAORAL ESCAPE<br />

(bpm) Pudding Liquid Solid<br />

A P A P A P<br />

Less than <strong>17</strong> 7 (37.0%) - 7 (37.0%) - 7 (37.0%) -<br />

<strong>17</strong>–18 8 (42.5%) 1 (5.0%) 6 (31.5%) 3 (16.0%) 6 (31.5%) 3 (16.0%)<br />

19 or more 2 (10.5%) 1 (5.0%) 3 (16.0%) - 1 (5.0%) 2 (10.5%)<br />

p 0.3860 0.2270 0.0681<br />

RR = respiratory rate, bpm = breaths per minute, A = absent, P = present.<br />

Fisher’s test at a significance level of 0.05.<br />

Table 4. Relationship of oxygen saturation with intraoral escape.<br />

O 2<br />

SATURATION<br />

INTRAORAL ESCAPE<br />

(mmHg) Pudding Liquid Solid<br />

A P A P A P<br />

Less than 80 1 (5.0%) 1 (5.0%) 2 (10.5%) - 1 (5.0%) 1 (5.0%)<br />

80–89 2 (10.5%) - 2 (10.5%) - 1 (5.0%) 1 (5.0%)<br />

90 or more 14 (73.5%) 1 (5.0%) 12 (64.0%) 3 (16.0%) 12 (64.0%) 3 (16.0%)<br />

p 0.3860 0.4696 0.2722<br />

O 2<br />

= oxygen, A = absent, P = present.<br />

Fisher’s test at a significance level of 0.05.<br />

Table 5. Relationship between smokers and non-smokers and intraoral escape.<br />

GROUP<br />

INTRAORAL ESCAPE<br />

Pudding Liquid Solid<br />

A P A P A P<br />

TAB 12 (63.5%) 2 (10.5%) 12 (65.0%) 2 (10.0%) 10 (55.0%) 4 (20.0%)<br />

NTAB 5 (26.0%) - 4 (20.0%) 1 (5.0%) 4 (20.0%) 1 (5.0%)<br />

p 0.5322 0.6244 0.6026<br />

TAB = smoker, NTAB = non-smoker, A = absent, P = present.<br />

Fisher’s test at a significance level of 0.05.<br />

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Macri et al.<br />

prevention of complications resulting from this disease,<br />

assisting in the control of morbidity and mortality and cost<br />

reduction (16).<br />

When the results of a previous study were corrected<br />

for smoking, the risk for chronic bronchitis became equal<br />

for men and women (<strong>17</strong>). In the current study of 19<br />

patients, 60.0% were male and 73.5% had contact with<br />

smoke or were smokers, which is similar to results found in<br />

the literature.<br />

Dyspnea is the most common symptom of COPD,<br />

and is a term used to characterize the subjective experience<br />

of respiratory distress. There is a correlation between<br />

COPD and swallowing disorders, which shows that<br />

breathing during swallowing is halted and resumed<br />

predominantly in the inspiratory phase, increasing the<br />

risk of aspiration (18). Cough can be considered as a<br />

variable parameter in clinical evaluation, since it can be<br />

present in both COPD and dysphagia. The presence of<br />

this symptom in patients with oropharyngeal dysphagia,<br />

regardless of form or intensity, is a warning sign of the<br />

presence of potential tracheal aspiration, especially when<br />

it is associated with food. This kind of aspiration increases<br />

the risk of aspiration pneumonia, regardless of the<br />

mechanism of action.<br />

In the present study, posterior intraoral escape was<br />

the variable most commonly found in patients during FEES.<br />

This finding is related to motor impairment of swallowing,<br />

and the longer the delay in firing the swallowing reflex, the<br />

greater the chance of aspirating part of the bolus, since the<br />

airway remains open (19,20).<br />

The presence of food residues in the epiglottic<br />

vallecula and/or piriform sinus can occur because of changes<br />

in the preparatory phase and/or oral swallowing, inefficiency<br />

of the ejection bolus, delayed triggering of the swallowing<br />

reflex, a decrease in peristalsis, reduced laryngeal elevation,<br />

or anterior and/or incoordination of the cricopharyngeal<br />

muscle (21).<br />

Since the presence of the characteristic symptoms<br />

of COPD affects the quality of life of these patients, we<br />

chose to correlate symptoms including degree of COPD,<br />

heart and respiratory rate, oxygen saturation, and smoking<br />

with intraoral exhaust with a view to subsequent observation<br />

of the impact of swallowing on worsening of pulmonary<br />

symptoms. Patients with COPD have impaired swallowing<br />

and a greater risk of aspiration pneumonia when compared<br />

with healthy patients (22).<br />

Patients with COPD have abnormal autonomic control<br />

of cardiac function and this study corroborates the research<br />

in question (23).<br />

The lack of coordination between swallowing and<br />

breathing increases the risk of aspiration in tachypneic or<br />

dyspneic patients, since they may not be able to tolerate<br />

longer periods or even short periods of apnea during<br />

swallowing. The normal respiratory rate based on a normal<br />

adult ranges from 12 to 20 breaths per minute (24).<br />

In the current study, it was observed that for<br />

pudding and liquid food consistencies, 10.5% of patients<br />

had posterior intraoral escape, but for solid food consistency,<br />

21.0% of patients had subsequent intraoral escape. There<br />

have been no previous studies on this association published<br />

in the literature. When cough is a major symptom during<br />

clinical evaluation of patients with COPD, it is evident that<br />

instrumental assessment is important for a more accurate<br />

diagnosis. With the evolution of disease, patients with<br />

COPD develop a profile of dysphagia, and the cough reflex<br />

should be taken into consideration by the audiologist as a<br />

possible sign of swallowing disorders (12).<br />

The role of the speech therapist in multidisciplinary<br />

teams is effective in dysphagic patient outcomes, and their<br />

early intervention allows regress of the increased risk of<br />

dysphagia in patients with COPD. The late identification of<br />

swallowing disorders can lead to severe pulmonary<br />

complications associated with aspiration (25).<br />

With health care programs aimed at recognition by<br />

professionals involved in the care of patients with COPD,<br />

the signs and symptoms of swallowing disorders can be<br />

incorporated into multidisciplinary care.<br />

CONCLUSION<br />

In this study, it was concluded that patients with<br />

COPD showed no oropharyngeal dysphagia upon clinical<br />

evaluation of swallowing. FEES revealed that there was a<br />

prevalence of oral dysphagia with the finding of subsequent<br />

intraoral escape. There was no relationship between<br />

subsequent intraoral escape and the degree of COPD,<br />

heart and respiratory rate, oxygen saturation, or smoking.<br />

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(gold) workshop summary. Am J. Respir. Crit. Care Med.<br />

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2. Menezes A, Perez-Padilla R, Jardim Jr, et al. Chronic<br />

obstructive pulmonary disease in five Latin American cities<br />

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(the PLATINO study): a prevalence study. Lancet.<br />

2005;366(9500):1875-81.<br />

3. Mannino DM, Holguin F. Epidemiology and global impact<br />

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Care Med. 2005;26(2):204-10.<br />

4. Campos HS. O preço da DPOC. Pulmão RJ. 2003;12(4):<br />

5-7.<br />

5. Mueller D. Terapia clínica nutricional na doença pulmonar.<br />

In: Mahan LK, Stump SE. Krause - Alimentos, nutrição e<br />

dietoterapia. 10th ed. São Paulo: Roca; 2002. p. 789-805.<br />

6. American Speech and Hearing Association (ASHA) (2004)<br />

[internet]. Model medical review guidelines for dysphagia<br />

services [monograph on the Internet]. [Revision to DynCorp<br />

2001 FTRP by ASHA]. [cited 2007 Mar 3]. Available from:<br />

http://www.asha.<strong>org</strong>/NR/rdonlyres/5771B0F7<br />

D7C04D47832A86FC6FEC2AE0/0/DynCorpDysph<br />

7. Michou E, Hamdy S. Cortical input in control of<br />

swallowing. Curr Opin Otolaryngol Head Neck Surg.<br />

2009;<strong>17</strong>(3):166-71.<br />

8. Rabe KF, Hurd S, Anzueto A, et al. Global Initiative for<br />

Chronic Obstructive Lung Disease: Global strategy for the<br />

diagnosis, management and prevention of chronic<br />

obstructive pulmonary disease: GOLD executive summary.<br />

Am J Respir Crit Care Med. 2007;<strong>17</strong>6(6):532-55.<br />

9. Wedzicha JA, Seemungal TA. COPDexacerbations:<br />

defining their cause and prevention. Lancet.<br />

2007;370(9589):786-96.<br />

10. Langmore S, Schatz K, Olsen N. Fiberoptic endoscopic<br />

examination of swallowing safety: a new procedure.<br />

Dysphagia. 1988;2(4):216-9.<br />

11. Global strategy for the diagnosis, management, and<br />

prevention of chronic obstructive pulmonary disease:<br />

Executive summary 2006. Global Initiative for Chronic<br />

Obstructive Lung Disease (GOLD) [Internet]. Available from<br />

http://www.goldcopd.<strong>org</strong>.<br />

12. Marik P. Aspiration pneumonitis and aspiration<br />

pneumonia. N Engl J Med. 2001;344(9):665-71.<br />

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misinformation: position of ADA. J. Amer Diet Assoc.<br />

2002;102(2):260-6.<br />

14. Crary M, Mann G, Groher M. Initial psychometric<br />

assessment of a functional oral intake scale for dysphagia<br />

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15. Rosenbek JC, Robbins J, Roecker EB, Coyle JL, Wood<br />

JL. A penetration aspiration scale. Dysphagia.<br />

1996;11(2):93-8.<br />

16. Skoretz S, Rebeyka D. Dysphagia following<br />

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Nurs. 2009;19(2):10-6.<br />

<strong>17</strong>. Menezes AM, Victora CG, Rigatto M. Prevalence and<br />

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18. Shaker R, Li Q, Ren J, et al. Coordination of deglutition<br />

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1992;263(5):750-5.<br />

19. Langmore SE, Schatz K, Olson N. Endoscopic and<br />

videofluoroscopic evaluations of swallowing and aspiration.<br />

Ann Otol Rhinol Laryngol. 1991;100(8):678-81.<br />

20. Helfrich-Miller KR, Rector KL, Straka JA. Dysphagia: its<br />

treatment in the profoundly retarded patient with cerebral<br />

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21. Costa M, Moscovici M, Pereira AA, Koch HA. A avaliação<br />

videofluoroscópica da transição faringoesofágica (esfíncter<br />

superior do esôfago). Radiol. Bras. 1993;26(2):71-80.<br />

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23. Paschoal MA, Petrelluzzi KF, Gonçalves NV. Estudo da<br />

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2002;11(1):27-37.<br />

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and outcome after acute stroke: does dysphagia matter?<br />

Stroke. 1996;27(7):1200-4.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):279-284.<br />

DOI: 10.7162/S1809-97772013000300008<br />

Characteristics of polypoid lesions in patients undergoing microsurgery of<br />

the larynx<br />

J<strong>org</strong>e Massaaki Ido Filho 1 , Bettina Carvalho 2 , Flavio Massao Mizoguchi 3 , Guilherme Simas do Amaral Catani 1 ,<br />

Evaldo Dacheux de Macedo Filho 1 , Osvaldo Malafaia 4 , Henrique J<strong>org</strong>e Stahlke Jr. 5<br />

1) ENT doctor recognized by ABORL (Physician at Hospital IPO and at the ENT Department of HC/UFPR.)<br />

2) ENT doctor recognized by ABORL (Fellow at Hospital IPO)<br />

3) ENT doctor recognized by ABORL (Physician at Hospital IPO)<br />

4) PhD in Medical Anatomy (1976). Professor associated with the postgraduate program in Surgical Principles at the Faculdade Evangelica do Parana.<br />

5) PhD in Clinical Surgery in the postgraduate program at UFPR (2002).Professor and Coordinator of Vascular Surgery at HC/UFPR.<br />

Institution: Hospital IPO.<br />

Curitiba / PR – Brazil.<br />

Mailin address: Hospital IPO - Dra Bettina Carvalho - Av. República Argentina, 2069 - Água Verde - Curitiba / PR – Brazil - Zip Code: 80620-010 - Telephone:<br />

(+55 41) 3314-1500 - http://ipo.com.br - E-mail: contato@ipo.com.br<br />

Article received on January 24 th , 2013. Article accepted on March 19 th , 2013.<br />

SUMMARY<br />

Introduction: Dysphonia is the main symptom of lesions that affect the vocal tract. Many of those lesions may require surgical<br />

treatment. Polyps are one of the most common forms of vocal cord lesions and the most prevalent indication for laryngeal<br />

microsurgery. There are different types of polyps, and their different characteristics can indicate different prognosis and treatments.<br />

Aim: To conduct a comparative study of polypoid lesions (angiomatous and gelatinous) in patients undergoing laryngeal<br />

microsurgery via an electronic protocol.<br />

Method: We prospectively evaluated 93 patients diagnosed with vocal fold polyps; the polyps were classified as angiomatous<br />

or gelatinous.<br />

Results: In total, 93 patients undergoing laryngeal microsurgery were diagnosed with vocal fold polyps. Of these, 63 (64.74%)<br />

had angiomatous and 30 (32.26%) gelatinous polyps. Most patients with angiomatous polyps were men; their polyps were<br />

frequently of medium size, positioned in the middle third of the vocal fold, and accompanied by minimal structural alterations<br />

(MSA). In contrast, the majority of patients with gelatinous polyps were women; their polyps were smaller, positioned in the<br />

middle and posterior third of the vocal fold, and were not accompanied by MSA. Both types of polyps were more frequently<br />

located on the right vocal fold.<br />

Conclusion: Angiomatous polyps were more frequently encountered than gelatinous polyps. In addition, correlations between<br />

polyp type and sex, polyp size, position, location, and the presence of MSA were observed. Different surgical techniques were<br />

used, but the postoperative results were similar and satisfactory after speech therapy.<br />

Keywords: Polyps; Vocal Cords; Microsurgery.<br />

INTRODUCTION<br />

Dysphonia is the main symptom of lesions that<br />

affect the vocal tract. More than 50% of individuals with<br />

voice disorders have benign alterations of the vocal fold<br />

mucosa. Polyps are one of the most frequent vocal cord<br />

lesions and are the most prevalent indication for laryngeal<br />

microsurgery. Like nodules, polyps are caused by overuse<br />

and abuse of the voice (1), although they may also occur<br />

as a result of a single traumatic incident (2,3). Trauma can<br />

affect the superficial vases of the lamina propria causing<br />

the liquids within to overflow, displacing the epithelial<br />

layer and inducing scarring due to the deposition of fibrin<br />

and vascular proliferation. Depending on the type of<br />

vascularization, polyps are classified as angiomatous or<br />

hemorrhagic if accompanied by vascularity or as hyaline or<br />

gelatinous in the absence of vascularization.<br />

The clinical picture is characterized by dysphonia<br />

related to intense vocal use, and is generally well-defined<br />

and recognized by the patient. The dysphonia is constant,<br />

and may progressively worsen. The voice presents as<br />

hoarse and breathy; sometimes it can be rough and,<br />

infrequently, diplophonic. A diagnosis is made by assessing<br />

the clinical history, and by perceptive analysis and<br />

observation of the phonatory system, which includes<br />

assessments of the phonatory posture adopted, articulation,<br />

and attitude. Laryngoscopy with or without stroboscopy<br />

can confirm the diagnosis. Treatment of vocal polyps is<br />

essentially surgical (4).<br />

Vocal polyps can have many presentations and<br />

characteristics, and the aim of our study was to identify the<br />

characteristics of the polyps found in our population.<br />

The objective of this study was to analyze and<br />

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Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx.<br />

Ido Filho et al.<br />

compare the features of polyps (intrinsic characteristics,<br />

associated lesions, and treatment outcomes) from patients<br />

undergoing laryngeal surgery at our hospital.<br />

METHODS<br />

This prospective study was conducted from February<br />

2010 to February 2011 using an electronic computerized<br />

protocol once the approval of the ethics committee had<br />

been obtained (CAEE :0286.0208.000-11). We used the<br />

SINPE © electronic protocol with the SINPE© Analyzer for<br />

data analysis (5,6,7). The protocol-based software program<br />

is capable of storing and manipulating data on a theoretical<br />

basis. The SINPE© Analyzer module is used to create reports,<br />

graphs, and statistics summarizing the main findings. A<br />

specific protocol for laryngeal disorders among the master<br />

ENT protocol available in SINPE was used for the analysis;<br />

only patients diagnosed with vocal fold polyps were analyzed.<br />

In total, 245 microsurgeries of the larynx were<br />

performed in our Hospital during the study period. The<br />

inclusion criteria were as follows: a diagnosis of polyps,<br />

clinical laryngoscopy, and intraoperative confirmation of<br />

the diagnosis by anatomopathology. Exclusion criteria<br />

were as follows: a diagnosis of infiltrative processes or<br />

storage disease (vocal polyps not identified upon<br />

anatomopathologic examination).<br />

Of the 245 patients who underwent surgery during<br />

the study period, 93 (36.61% of lesions) with vocal polyps<br />

and an indication for microsurgery were evaluated. These<br />

93 patients were classified into 2 groups according to the<br />

physical and histological characteristics of the lesions: (a)<br />

those with angiomatous polyps (n = 63, 67.75%) and (b)<br />

those with gelatinous polyps (n = 30, 32.25%). Comparisons<br />

between the 2 groups were made according to 12 anatomic<br />

and surgical parameters based on the polyp characteristics.<br />

Parameters 1–8 refer to the intrinsic characteristics of the<br />

polyps (Table 1), parameters 9 and 10 refer to polypassociated<br />

lesions (Table 2), and parameters 11 and 12<br />

refer to the treatment strategy (Table 3).<br />

Table 1. Polyp Intrinsic Characteristics.<br />

Parameter Description<br />

1. Sex male or female<br />

2. Age<br />

3. Size a) small: the polyp takes up one-third of the vocal fold<br />

b) medium: the polyp occupies two-thirds of the vocal fold<br />

c) large: the polyp fills more than two-thirds of the vocal fold<br />

4. Implantation a) sessile: the lesion is considered sessile if it has a wide base<br />

b) pedunculated: the polyp presenting small implantation with elevation of its base (with a stalk or peduncle)<br />

5. Lobulation a) unilobulated: only one lobulation<br />

b) bilobulated: two lobulations<br />

c) multilobulated: many lobulations<br />

6. Position a) anterior third: the polyp is within the anterior third of the vocal fold<br />

b) middle third: the polyp is within the middle third of the vocal fold<br />

c) posterior third: the polyp is within the posterior third of the vocal fold<br />

7. Location a) free edge: the polyp is located at the free edge of the vocal fold<br />

b) upper edge: the polyp is located at the top edge of the vocal fold<br />

c) lower edge: the polyp is located on the bottom edge of the vocal fold<br />

d) more than one location: the polyp covers various sites (e.g., transglottic polyp)<br />

8. Location of polyp a) right vocal fold<br />

on vocal fold b) left vocal cord<br />

c) bilateral: polyp on both the right and left folds<br />

Table 2. Associated lesions.<br />

Parameter<br />

Description<br />

9. Presence of associated lesions and their type a) nodular reaction d) groove bag g) mucous cyst<br />

b) varicosity e) stria minor h) mucosa bridge<br />

c) stria major f) intracordal cyst i) microdiaphragm<br />

10. Associated lesions in relation to the vocal fold a) ipsilateral - on the same vocal fold polyp<br />

b) contralateral - on opposite vocal fold polyp<br />

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Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx.<br />

Ido Filho et al.<br />

Table 3. Treatment.<br />

Parameter Description<br />

11. Surgical technique a) polyp grip + microscissors (holding the polyp and performing resection with microscissors)<br />

b) medial microflap + microscissors (microbisturi incision, creation of a medial microflap, and resection with<br />

microscissors)<br />

12. Evolution of a)optimal: satisfactory treatment<br />

Speech therapy b) poor: unsatisfactory treatment<br />

Table 4. Comparative analysis of internal characteristics of angiomatous and gelatinous polyps.<br />

Parameter Angiomatous Gelatinous p < 0.05<br />

n = 63 (64.74%) n = 30 (32.26%)<br />

1. Sex<br />

Male 41 (65.08%) 10 (33.33%)<br />

Female 22 (34.92%) 20 (66.67%) p = 0.008<br />

2. Age 41–60 years (49.21%) 20–40 years (46.67%)<br />

3. Size Medium Small<br />

43 (68.25%) <strong>17</strong> (56.67%) p = 0.001<br />

4. Implantation Pedunculated Sessile<br />

35 (55.56%) 19 (63.33%)<br />

5. Lobulation Unilobulated Unilobulated<br />

53 (84.13%) 26 (86.67%)<br />

6. Position<br />

anterior third 25 (35.71%) 8 (26.67%)<br />

middle third 36 (51.43%) 11 (36.67%) p = 0.02<br />

posterior third 9 (12.86%) 11 (36.67%)<br />

*Number of positions: 34 (53.96%) 24 (80%) p = 0.03<br />

One 29 (46,04%) 6 (20%)<br />

More than one<br />

7. Location Free edge Free edge<br />

42 (66.67%) 20 (66.67%)<br />

8. Location of polyp on<br />

vocal fold:<br />

right 39 (61.9%) 14 (46.67%)<br />

left 22 (34.92%) 9 (30%) p = 0.009<br />

bilateral 2 (3.<strong>17</strong>%) 7 (23.33%)<br />

The protocols were performed on the day before<br />

surgery after the pre-anesthetic consultation, and<br />

supplemented during the postoperative follow-up.<br />

Endolaryngeal microsurgeries were performed in the<br />

operating room of the same institution using suspension<br />

laryngoscopy (SL) by 3 doctors from the Laryngology and<br />

Voice Service department.<br />

During SL, the vocal folds were subject to visual<br />

inspection and palpation with delicate microforceps. The<br />

microsurgical technique used depended on the preoperative<br />

evaluation and its confirmation during surgery.<br />

Statistical analyses were performed using the Chisquare<br />

test to compare the variables discussed above, and<br />

the significance level was set at p < 0.05.<br />

RESULTS<br />

All 93 patients underwent laryngeal surgery due to<br />

a diagnosis of polyps of the vocal folds during the period<br />

February 2010 to February 2011. Of these, 63 (64.74%)<br />

had angiomatous and 30 (32.26%) had gelatinous polyps.<br />

Regarding sex, 51 were male and 42 female. Their age<br />

ranged from 20 to 80 years. The distribution according to<br />

the 12 parameters for each type of polyp is shown in<br />

Tables 4, 5, and 6.<br />

DISCUSSION<br />

Benign lesions of the vocal folds represent a<br />

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Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx.<br />

Ido Filho et al.<br />

Table 5. Comparative analysis of parameters relating to lesions associated with angiomatous and<br />

gelatinous polyps.<br />

Parameter Angiomatous Gelatinous p < 0.05<br />

n = 63 (64.74%) n = 30 (32.26%)<br />

9. Associated lesions p = 0.02<br />

a) nodular reaction 26 (44.83%) 13 (65%)<br />

b) varicosity 8 (13.79%) 4 (20%)<br />

c) stria major 8 (13.79%) 1 (5%)<br />

d) groove bag 4 (6.9%) 0<br />

e) stria minor 3 (5.<strong>17</strong>%) 0<br />

f) intracordal cyst 4 (6.9%) 1 (5%)<br />

g) mucous cyst 2 (3.45%) 1 (5%)<br />

h) mucosa bridge 2 (3,45%) 0<br />

i) microdiaphragm 1 (1.72%) 0<br />

* Presence of MSA<br />

Present 30 (47,61%) 6 (20%)<br />

Absent 33 (52.39%) 24 (80%)<br />

10. Association of lesions<br />

with the vocal fold:<br />

Ipsilateral 44 (69.84%) 27 (76.67%)<br />

Contralateral 19 (30.16%) 7 (23.33%)<br />

Table 6. Comparative analysis of parameters for treatment of angiomatous and gelatinous polyps.<br />

Parameter Angiomatous Gelatinous p < 0.05<br />

n = 63 (64.74%) n = 30 (32.26%)<br />

11. Surgical technique<br />

Grip+ microscissors 36 (57.14%) 12 (40%)<br />

Medial microflap+ microscissors 27 (42.6%) 18 (60%)<br />

12. Evolution of speech therapy Optimal Optimal<br />

56 (88.89%) 28 (93.33%)<br />

significant problem for otolaryngologists because they are<br />

very common and require a multidisciplinary treatment<br />

approach. When such lesions do not respond to drug<br />

therapy and/or speech therapy, surgery is required with<br />

the aim of increasing phonatory function or establishing a<br />

histological diagnosis by biopsy (8, 9).<br />

In our study, we found 93 (36.61%) polyps among<br />

245 patients with vocal fold lesions who underwent laryngeal<br />

surgery during the period from February 2010 to February<br />

2011 in our Hospital. This was the most frequent diagnosis<br />

observed in this study, which supports the reports by Haas<br />

& Doderlein, Mossallam, and Lehmann and Kleinsasser,<br />

who noted that polyps were the main indication for<br />

laryngeal microsurgery (10-13).<br />

Angiomatous polyps were more common (63<br />

patients, 64.74%) than gelatinous polyps (30 patients,<br />

32.26%). In addition, we noticed a male predominance<br />

among patients with angiomatous polyps and a female<br />

predominance among those with gelatinous polyps. The<br />

Chi-square test (significance level, p = 0.008) showed that<br />

the type of polyp was dependent on sex. The male<br />

predominance of angiomatous polyps (65.08%) is<br />

consistent with the findings reported in the literature<br />

(3,14,15), but there was no male predominance of<br />

gelatinous polyps. Instead, these were more common in<br />

women (66.67%), which is a similar finding to that<br />

obtained by Dailey, who reported a female incidence of<br />

62% among patients with gelatinous polyps (16). This<br />

may be because women are more likely to seek medical<br />

advice than men. In addition, female patients with<br />

increased vocal fold mass have a lower pitch, which has<br />

a greater impact on the social life of these patients than<br />

is the case with male patients.<br />

Regarding size, the statistical analysis demonstrated<br />

that the polyp size was dependent on type. Most<br />

angiomatous polyps were of medium size (68.25%) whereas<br />

most gelatinous polyps were small (56.67%) (p = 0.001).<br />

Angiomatous polyps were also found most frequently in<br />

the middle third of the vocal fold (51.43%), whereas the<br />

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Characteristics of polypoid lesions in patients undergoing microsurgery of the larynx.<br />

Ido Filho et al.<br />

gelatinous polyps tended to be found in the middle and<br />

posterior thirds of the fold (36.67% each) (p = 0.02). Thus,<br />

the position of the polyp in the vocal fold was also<br />

dependent on type.<br />

In our study, both angiomatous and gelatinous<br />

polyps were predominantly found on the right vocal fold<br />

(p = 0.009), and so there is also a relationship between<br />

polyp type and its location on the vocal fold. Eckley et al.<br />

reported similar findings (14), whereas Sakae et al. (<strong>17</strong>)<br />

reported that polyps were found in the left vocal fold in<br />

53% of cases.<br />

The presence of MSA occurred in 47.61% of patients<br />

with angiomatous polyps, in comparison with 20% of<br />

patients with gelatinous polyps (p = 0.02). There was thus<br />

also a correlation between polyp type and the presence of<br />

MSA (greater in angiomatous polyps). In Sakae et al.’s<br />

study (16), polyps were associated with MSA in 23.5% of<br />

patients.<br />

Reaction nodular lesions were found in 41.26%<br />

of the patients with angiomatous polyps and 43.33% of<br />

the patients with gelatinous polyps. There was no<br />

statistical difference between the two groups (p =<br />

0.97). In Eckley et al.’s study (14), 37% of patients had<br />

reaction lesions, confirming the suspicion that the<br />

impact of the polyp on the healthy vocal fold can cause<br />

long-term alterations in the epithelial layer of the<br />

contralateral vocal fold (14,18).<br />

Regarding the MSA, stria were the most frequently<br />

encountered lesions (49.9%) in angiomatous polyps,<br />

whereas varicosity was predominant in gelatinous polyps<br />

(66.66%). In Eckley et al.’s study (14), stria were the most<br />

frequently encountered MSA (70%).<br />

In our study, the patients underwent larynx surgery<br />

with SL, which led to considerable improvement in voice<br />

quality and the remission of other symptoms. The surgical<br />

technique most commonly used for angiomatous polyps is<br />

to grab the polyp and resect it with a microscissors<br />

(57.14%). For gelatinous polyps, the main surgical technique<br />

is to create a medial microflap and then resect it with a<br />

microscissors (60%). There was no difference in outcome<br />

when these techniques were compared.<br />

After surgery, all of the patients underwent speech<br />

therapy with optimal developments in most cases (88.89<br />

and 93.33% for those with angiomatous and gelatinous<br />

polyps, respectively). There is a consensus regarding the<br />

use of postoperative speech therapy and appropriate<br />

follow-up and successful outcome (9). We plan to undertake<br />

further analysis of the therapy outcomes in subsequent<br />

research projects.<br />

CONCLUSION<br />

In this study, angiomatous polyps were more<br />

frequently encountered than gelatinous polyps. There was<br />

a male predominance among patients with angiomatous<br />

polyps, and a female predominance among those with<br />

gelatinous polyps. Angiomatous polyps were more<br />

frequently associated with MSA. There was also a correlation<br />

between the size of the polyp, and its position and location<br />

in the vocal fold and the presence of MSA. Different<br />

surgical techniques were used, but the postoperative<br />

results were similar and satisfactory once the therapy was<br />

complete.<br />

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o Sistema Integrado de Protocolos Eletrônicos [dissertation].<br />

[Curitiba]: Universidade Federal do Paraná; 2005. 111 p.<br />

7. Catani GSA, Carvalho B, Ido Filho JM, Macedo Filho ED,<br />

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protocol for epidemiological study of patients undergoing<br />

microsurgery of the larynx. Int. Arch. Otorhinolaryngol.<br />

2012;16(3):346-52.<br />

8. Woo P, Noordzij JP. Glottal area waveform analysis of<br />

benign vocal fold lesions before and after surgery. Ann Otol<br />

Rhinol Laryngol. 2000;109:441-6.<br />

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FV, Rapoport P. Pólipos de pregas vocais: aspectos clínicos<br />

e cirúrgicos. Rev Bras. Otorrinolaringol. 2002;68(4):534-<br />

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Laryngologie, Rhinologie, Otologie. 1978;57(3):235-42.<br />

11. Mossallam I, Kotby MN, Ghaly AFEA. Histopathological<br />

aspects of benign vocal fold lesionsassociated with<br />

dysphonia. In: Kirchner JA, editor. Vocal Fold histipathology:<br />

A symposium. San Diego: College-Hill; 1986. p. 65-80.<br />

12. Lehmann W, Pampurik J, Guyot JP. Laryngeal pathologies<br />

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1974;22(3):69-83.<br />

14. Eckley CA, Swensson J, Duprat AC, Donati F, Costa HO.<br />

Incidência de alterações estruturais das pregas vocais<br />

associadas ao pólipo de prega vocal. Rev Bras<br />

Otorrinolaringol. 2008:74(4):508-11.<br />

15. Neves BM, Neto JG, Pontes P. Diferenciação<br />

histopatológica e himunoistoquímica das alterações epiteliais<br />

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2007;21(1):112-8.<br />

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2004:70(6):743-8.<br />

18. Marcotullio D, Magliulo G, Pietrunti S, Suriano M.<br />

Exudative laryngeal diseases of Reinke’s space: a<br />

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2002;31:376-80.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):285-290.<br />

DOI: 10.7162/S1809-97772013000300009<br />

Audiological outcomes of cochlear implantation in Waardenburg Syndrome<br />

Ana Tereza de Matos Magalhães 1 , Paola Angélica Samuel 1 , Maria Valeria Schimdt Goffi-Gomez 2 , Robinson Koji Tsuji 3 ,<br />

Rubens Brito 3 , Ricardo Ferreira Bento 3 .<br />

1) Audiologist. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

2) Audiologist, PhD. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

3) MD, PHD. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

Institution: Department of Otolaryngology, University of São Paulo School of Medicine.<br />

São Paulo / SP - Brazil.<br />

Mailing address: Ana Tereza de Matos Magalhães - Rua Capote Valente, 432 conjunto 14 - São Paulo / SP - Brazil - Zip code: 05409-001 - E-mail: anatereza@forl.<strong>org</strong>.br<br />

Article received on February 11 th , 2013. Article accepted on April 23 th , 2013.<br />

SUMMARY<br />

Introduction: The most relevant clinical symptom in Waardenburg syndrome is profound bilateral sensorioneural hearing loss.<br />

Aim: To characterize and describe hearing outcomes after cochlear implantation in patients with Waardenburg syndrome to<br />

improve preoperative expectations.<br />

Method: This was an observational and retrospective study of a series of cases. Children who were diagnosed with Waardenburg<br />

syndrome and who received a multichannel cochlear implant between March 1999 and July 2012 were included in the study.<br />

Intraoperative neural response telemetry, hearing evaluation, speech perception, and speech production data before and after<br />

surgery were assessed.<br />

Results: During this period, 806 patients received a cochlear implant and 10 of these (1.2%) were diagnosed with Waardenburg<br />

syndrome. Eight of the children received a Nucleus 24 ® implant and 1 child and 1 adult received a DigiSonic SP implant. The<br />

mean age at implantation was 44 months among the children. The average duration of use of a cochlear implant at the time<br />

of the study was 43 months. Intraoperative neural responses were present in all cases. Patients who could use the speech<br />

processor effectively had a pure tone average of 31 dB in free-field conditions. In addition, the MUSS and MAIS questionnaires<br />

revealed improvements in speech perception and production. Four patients did not have a good outcome, which might have<br />

been associated with ineffective use of the speech processor.<br />

Conclusion: Despite the heterogeneity of the group, patients with Waardenburg syndrome who received cochlear implants<br />

were found to have hearing thresholds that allowed access to speech sounds. However, patients who received early intervention<br />

and rehabilitation showed better evolution of auditory perception.<br />

Keywords: Hearing; Cochlear Implants; Hearing Loss; Waardenburg Syndrome; Speech Perception.<br />

INTRODUCTION<br />

Cochlear implantation outcomes are dependent on<br />

several factors that lead to a good prognosis, such as early<br />

diagnosis and intervention, systematic rehabilitation, family<br />

permeability, and type of communication, as well as other<br />

factors related to hearing loss including etiology, period of<br />

deafness, and the presence of other associated impairments<br />

(1-3).<br />

Genetic causes of deafness include Waardenburg<br />

syndrome (WS), which accounts for 2%–5% of patients<br />

with congenital hearing loss (4). WS is an autosomal<br />

dominant disease characterized by hyperplasia of the<br />

medial portion of the eyebrows, a broad nasal root,<br />

heterochromia iris, white forelock or early graying, and<br />

congenital sensorineural hearing loss (5).<br />

The most relevant clinical symptom in WS is the<br />

hearing loss, which can be unilateral or bilateral, and<br />

moderate to profound. Severe to profound deafness is<br />

more evident in patients with Type I and Type II WS, with<br />

an incidence of 35%–75% and 55%–91%, respectively<br />

(6). Cochlear implantation is indicated for patients with<br />

bilateral and severe to profound hearing loss who are<br />

unable to benefit from conventional hearing aids.<br />

Few studies have evaluated the audiological<br />

outcomes of cochlear implantation in users with syndromes.<br />

Some have noted limited success depending on the<br />

characteristics associated with the syndrome and the age at<br />

implantation. Studies of WS have reported good outcomes<br />

for those with no other impairments associated with the<br />

syndrome (7-10).<br />

The purpose of this study was to characterize and<br />

describe hearing outcomes after cochlear implantation<br />

in patients with WS to improve preoperative<br />

expectations.<br />

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Audiological outcomes of cochlear implantation in Waardenburg Syndrome.<br />

Magalhães et al.<br />

METHOD<br />

This was a retrospective and observational study of<br />

a series of cases and was performed from March 1999<br />

through July 2012. Data wee collected from the cochlear<br />

implant team database. Patients who met the following<br />

criteria were included in the study:<br />

• Diagnosed with WS;<br />

• Multichannel cochlear implant user.<br />

The following data were collected:<br />

• Age at diagnosis of deafness;<br />

• Age at rehabilitation initiation;<br />

• Age at cochlear implant activation;<br />

• Duration of cochlear implant use (in months);<br />

• Number of active electrodes;<br />

• Intraoperative neural telemetry results;<br />

• Aided average pure tone threshold in sound field c<br />

before and after surgery;<br />

• Pre- and post-operative speech perception and<br />

production test results.<br />

Intraoperative neural telemetry data were recorded<br />

by Nucleus NRT 3.1 ® or Custom Sound EP 2.0 ® software<br />

from Cochlear Corporation ® . A response was considered<br />

present when at least 3 electrodes presented reproducible<br />

responses. Electrical Auditory Brainstem Response (EABR)<br />

data were recorded by Bio-logic v.7.0 ® and stimulated by<br />

Digistim ® from Neurelec ® .<br />

Free field audiometry data were collected using<br />

Madsen Midimate 622 ® and retrieved from patient records.<br />

Where no threshold was available (such as during preoperative<br />

assessment), a value of 130 dB was used to<br />

calculate the average (greater than the amplifier limit); the<br />

calculation was performed according to the BIAP<br />

recommendation (11).<br />

Speech perception and production were evaluated<br />

using our standard protocols and were assessed according<br />

to age (12). For children up to the age of 4 years 11 months,<br />

the Early Perception Test (ESP) was used (the Portuguese<br />

form by Orlandi and Bevilacqua (13)). For children aged 5<br />

years or older, the Portuguese form of the Glendonald<br />

Auditory Screening Procedure (GASP) was used (14). In<br />

both tests, the results were classified into the speech<br />

perception categories described by Geers (15).<br />

Two inventories were submitted to the parents, one<br />

addressing information on the frequency with which the<br />

child showed significant auditory behaviors in their everyday<br />

life (IT-MAIS - Infant-Toddler Meaningful Auditory<br />

Integration Scale) (16, <strong>17</strong>), the other providing information<br />

on the frequency with which the child demonstrated<br />

Table 1. Degree of permeability of the family and the cognitive<br />

style of the children.<br />

Score<br />

Classification<br />

90–100% Excellent<br />

60–89% Satisfactory<br />


Audiological outcomes of cochlear implantation in Waardenburg Syndrome.<br />

Magalhães et al.<br />

Table 2. Characteristics of cochlear implant patients.<br />

ID SEX* Implantation age Implanted ear Number of electrodes Intraoperative neural Brand of cochlear<br />

(months) activated telemetry implant **<br />

1. MLFN F 53 Right 22/22 Present N24<br />

2. JLCS M 20 Right 22/22 Present N24 RE<br />

3. GAS M 36 Right 22/22 Present N24<br />

4. MSLG M 18 Right 22/22 Present N24<br />

5. ASLG F 106 Left 22/22 Present N24<br />

6. AMNCT F 71 Right 22/22 Present N24 RE<br />

7. ACAFD F 38 Right 22/22 Present N24 RE<br />

8. VMM M 32 Right 22/22 Present N24 RE<br />

9. ERM M 30 Left 18/20 Present Digisonic<br />

10. AAC F 22 years Right 19/20 Present Digisonic<br />

*M = male; F = female; **N24 = Nucleus 24; N24RE = Nucleus 24 Freedom.<br />

Table 3. Data on hearing loss, rehabilitation, degree of permeability of the family, and the cognitive style of the children.<br />

ID Diag. Hearing aid Rehab. Rehabilitation Number of sess. Freq. Permeabil Cognitive<br />

(months) (months) (months) (per week) Style<br />

1. MLFN 6 15 12 Total Communicat. Once Inconsistent Low Satisfactory<br />

2. JLCS 10 16 12 Audio-oral Twice Consistent Excellent Satisfactory<br />

3. GAS 14 16 16 Audio-oral Twice Consistent Excellent Satisfactory<br />

4. MSLG 9 12 15 Audio-oral Twice Inconsistent Low Low<br />

5. ASLG 12 36 36 Audio-oral Twice Inconsistent Low Satisfactory<br />

6. AMNCT 20 24 24 Audio-oral Twice Consistent Low Satisfactory<br />

7. ACAFD 20 26 26 Audio-oral Twice Consistent Excellent Low<br />

8. VMM 5 18 12 Audio-oral Twice Consistent Satisfactory Satisfactory<br />

9. ERM 6 14 14 Audio-oral Twice Consistent Satisfactory Low<br />

10. AAC 7 24 24 Audio-oral Twice Consistent — —<br />

ID: identification; Diag: Age at diagnosis; Hearing aid: Age when began using hearing aids; Rehab.: Beginning of rehabilitation;<br />

sess.: sessions; Freq.: Frequency of rehabilitation; Permeabil: Degree of permeability of the family.<br />

Table 4. Audiological outcomes and speech perception among children with a cochlear implant.<br />

ID Effective use of CI Time of CI use PTA (em dBNA) Category IT-MAIS/MAIS MUSS<br />

Pre CI Pos CI Pre CI Pos CI Pre CI Pos CI Pre CI Pos CI<br />

1. MLFN Yes <strong>17</strong>4 110 25 0 5 25% 100% 20% 100%<br />

2. JLCS Yes 60 105 20 0 6 0% 100% 40% 97.5%<br />

3. GAS Yes 54 105 25 0 6 7.5% 100% 45% 100%<br />

4. MSLG No 48 100 60 0 1 30% <strong>17</strong>.5% 25% 2.5%<br />

5. ASLG No 36 70 50 0 2 55% 70% 80% 85%<br />

6. AMNCT No 24 75 50 0 1 0% 60% 0% 75%<br />

7. ACAFD No 6 ABS 110 0 0 10% 30% 40% 37%<br />

8. VMM YES 24 75 30 0 4 5% 75% 20% 80%<br />

9. ERM YES 6 ABS NR 0 1 0% 20% 0% 15%<br />

CI: cochlear implant; PTA (Pure tone average) Average threshold at 500 Hz and 4000 Hz (11); ABS = Absent; NR=unrealized;<br />

Category: Category of speech perception (15).<br />

MUSS and MAIS questionnaire responses did not show any<br />

improvement in speech perception or production.<br />

Case 10 was able to discriminate all of the vowels<br />

and 80% of a closed-set sentence after 9 months of using<br />

the speech processor.<br />

DISCUSSION<br />

All patients were diagnosed with severe to profound<br />

congenital hearing loss, which is in accordance with Barzotto<br />

and Folador (21), who showed that the most common form<br />

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Audiological outcomes of cochlear implantation in Waardenburg Syndrome.<br />

Magalhães et al.<br />

of hearing loss in WS is profound sensorineural. The<br />

prevalence of patients with WS who received a cochlear<br />

implant in this study was 1.2%, which is close to the value<br />

generally reported in the literature (8-10, 22).<br />

During the pre-implant evaluation, no patient was<br />

found to have benefited from conventional hearing aids,<br />

which corresponds to category 0 of speech perception and<br />

an inability to detect speech sounds. Thus, cochlear<br />

implantation was indicated.<br />

All patients had complete insertion of electrodes<br />

and showed an intraoperative neural response, which<br />

means that the auditory nerve responded to the first<br />

electrical stimulation of the cochlear implant. Guedes et<br />

al.(1) showed that adult patients who showed intraoperative<br />

telemetry responses had better results in speech perception<br />

tests, but this relationship was not statistically significant<br />

among children.<br />

Assessment of the hearing outcomes of all patients<br />

who were able to use the cochlear implant effectively<br />

showed that all had audiometric thresholds that enabled<br />

perception of speech sounds (according to the audiogram<br />

of Portuguese speech sounds) (23).<br />

The IT-MAIS questionnaire results also showed<br />

significant clinical improvements in most cases, reflecting<br />

improvements in listening skills, not only for detection, but<br />

also for the recognition of some sounds, since most of the<br />

patients had a good hearing threshold. Kubo et al. (3)<br />

showed that after 6 to 12 months of use of a cochlear<br />

implant, children were able to distinguish and recognize<br />

sounds. In cases 4, 5, and 7 in the present study no clinical<br />

improvement was detected by IT-MAIS because the child<br />

didn´t use the cochlear implant effectively, that is, there<br />

were care and maintenance problems as well as infrequent<br />

use of the implant during the rehabilitation process.<br />

The MUSS results, which reflect oral language skills,<br />

showed slow improvements. These skills are dependent<br />

on daily experience, systematic rehabilitation, and<br />

stimulation by the family according to Kobo et al. (3). In our<br />

study, we found that children with low family permeability<br />

did not show any difference in their MUSS responses after<br />

cochlear implantation. Cases 4 and 5 are brothers and the<br />

family was not involved with their therapy or the fitting of<br />

their speech processors. In case 7, the family stopped<br />

using the speech processor because the child did not seem<br />

to improve.<br />

These findings indicate that cochlear implants provide<br />

access to speech sounds, but that the development of<br />

auditory and language skills is dependent on systematic<br />

rehabilitation and family involvement (2).<br />

There are a few studies of cochlear implantation in<br />

patients with syndromes in the literature, and these show<br />

that patients who have no other associated intellectual<br />

impairments, who receive their implant early, and who are<br />

subject to sufficient stimulation have good outcomes (7-<br />

10, 24).<br />

In cases 2 and 3 in the present study, significant<br />

improvements in speech perception were observed and<br />

the family was also a meaningful participant in the<br />

therapeutic process. In case 1, however, although an<br />

improvement in hearing behavior was observed, the<br />

patient’s oral language was below average after<br />

implantation. The family showed a low level of participation<br />

in the patient’s rehabilitation process and there was<br />

inconsistent use of the implant owing to poor care of the<br />

equipment, which led to numerous maintenance and<br />

technical assistance events, and thus undermined the<br />

patient’s performance.<br />

Out of the 10 cases described here, 3 (cases 5, 6, and<br />

7) presented with late fitting of hearing aids, late auditory<br />

rehabilitation, and late implantation, which was reflected in<br />

their speech perception tests. Several studies have shown<br />

that children experience greater benefits from cochlear<br />

implantation when the implant is fitted when they are<br />

younger than age 2, which is the ideal period for better<br />

leveraging the outcomes of the cochlear implant (25, 26).<br />

These children may show development patterns similar to<br />

those of children with normal hearing (27).<br />

Andrade et al. (10) also confirmed that prelingually<br />

deafened WS children who have prior nonsignificant<br />

or marginal benefit from acoustic amplification<br />

but normal inner ear anatomy are potentially good<br />

candidates for audio-oral rehabilitation with a cochlear<br />

implant. Postoperative performance outcomes of 7 cases<br />

with WS were also assessed and compared to results<br />

obtained by children with non-syndromic congenital<br />

deafness. No statistical differences were found between<br />

the groups.<br />

Therefore, early intervention and rehabilitation is<br />

essential for children with WS as well the profoundly<br />

hearing impaired. This will ensure that they are offered<br />

better conditions to achieve good outcomes with a cochlear<br />

implant. Parental involvement throughout the rehabilitation<br />

process is also important for improving the quality of<br />

communication.<br />

Results from case 10 were not satisfactory despite<br />

having early intervention and childhood rehabilitation.<br />

On the other hand, the patient has not used the cochlear<br />

implant for very long, and these results may improve<br />

over time.<br />

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Magalhães et al.<br />

Genetic counseling is important for predicting the<br />

risk of transmission as well as for studying the family of the<br />

affected individual (21, 22), and genetic findings may<br />

influence the prognosis and treatment opportunities.<br />

CONCLUSION<br />

In our group of patients with WS who received a<br />

cochlear implant, hearing thresholds that allow access to<br />

speech sounds were achieved. However, those who showed<br />

good evolution of the perception of auditory and oral<br />

language skills were those who received early stimulation,<br />

systematic rehabilitation, and who had a family that was<br />

actively involved in the process.<br />

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Otolaryngol. 2008;128(4):373-7.<br />

26. Profant M, Kabatova Z, Simkova L. From hearing<br />

screening to cochlear implantation: cochlear implants in<br />

children. Acta Otolaryngol. 2008;128(4):369-72.<br />

27. Stuchi R, Nascimento L, Belivacqua M, Brito Neto R.<br />

Linguagem oral de crianças com cinco anos de uso do implante<br />

coclear. Pró-Fono R. Atual. Cient. 2007;19(2):167-76.<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 285-290, Jul/Aug/September - 2013.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):291-304.<br />

DOI: 10.7162/S1809-977720130003000010<br />

Performance analysis of ten brands of batteries for hearing aids<br />

Silvio Pires Penteado 1 , Ricardo Ferreira Bento 2 .<br />

1) Electronic Engineer, PhD in Medical Science. Department of Otorhinolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

2) PhD, Professor, Chairman. Department of Otorhinolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

Institution: Department of Otorhinolaryngology, University of São Paulo School of Medicine.<br />

São Paulo / SP – Brazil.<br />

Mailing address: Silvio Pires Penteado - Av. Dr. Enéas Carvalho de Aguiar 255, 6 th floor - São Paulo / SP – Brazil – ZIP code: 05403-000 - Telephone: (+55 11)<br />

2661-6539 - E-mail : penteadosp@gmail.com<br />

Financial Support: Fundação Otorrinolaringologia<br />

Article received on February 22 th , 2013. Article accepted on April 30 th , 2013.<br />

SUMMARY<br />

Introduction: Comparison of the performance of hearing instrument batteries from various manufacturers can enable otologists,<br />

audiologists, or final consumers to select the best products, maximizing the use of these materials.<br />

Aim: To analyze the performance of ten brands of batteries for hearing aids available in the Brazilian marketplace.<br />

Methods: Hearing aid batteries in four sizes were acquired from ten manufacturers and subjected to the same test conditions<br />

in an acoustic laboratory.<br />

Results: The results obtained in the laboratory contrasted with the values reported by manufacturers highlighted significant<br />

discrepancies, besides the fact that certain brands in certain sizes perform better on some tests, but does not indicate which<br />

brand is the best in all sizes.<br />

Conclusions: It was possible to investigate the performance of ten brands of hearing aid batteries and describe the procedures<br />

to be followed for leakage, accidental intake, and disposal.<br />

Keywords: Hearing Aids; Performance Tests; Benchmarking; Batteries; Reference Standards; Laboratory Equipment.<br />

INTRODUCTION<br />

Dillon (2000) posits that the history of the<br />

development of hearing aids can be divided into four eras:<br />

the acoustic, carbon valve, transistor, and digital ages. It was<br />

only during the first two of these eras that batteries were<br />

not needed to power electrical or electronic circuits.<br />

Lybarger (1988) states that the first powered hearing aid in<br />

the U.S. was produced by Miller Reese Hutchinson in 1902;<br />

later, some hearing aids with portable tubes and a drain<br />

current of 60 mA were manufactured. Lybarger describes<br />

that the evolution of tubes into transistors brought no<br />

significant change in the noise performance of hearing aids;<br />

it did, however, drastically reduce battery drain and the size<br />

of the batteries required (the drain current was decreased<br />

by about 100 times). Modern hearing aids use the topology<br />

of transistor analog hearing aids with other electronic<br />

circuits that result in devices with better performance and<br />

electroacoustic features, but higher power consumption<br />

(Cudahy and Levitt, 1994). Kates (2008) states that the<br />

digital processor, memory, and analog/digital converter<br />

(the internal circuits of the digital signal processor) are<br />

responsible for about 70% of the entire energy consumption<br />

of a digital hearing aid.<br />

|Hearing aid batteries based on mercury were<br />

gradually replaced by zinc-air batteries, providing positive<br />

effects on the environment and advantages for patients<br />

(Sparkes and Lacey, 1997). As described by Bloom (2003),<br />

“the tiny button cells used in contemporary hearing<br />

instruments typically have double the life of the old<br />

mercury cells and three times that of silver oxide” and they<br />

are “small, lightweight, and leak resistant, offering large<br />

capacity, stable voltage, and start-up on demand.”<br />

Knutsen (1982) describes a cell as a device for<br />

converting chemical energy into electrical energy, with a<br />

set amount of voltage and current necessary to power an<br />

electrical or electronic circuit. Bocchi, Ferracin, and Biaggio<br />

(2000) review the confusion arising from the terms pills<br />

and batteries, and state that while the former should be<br />

understood as a device consisting of two electrodes and an<br />

electrolyte, the second is an association of two or more<br />

pills.<br />

Pinkwart and Tuebke (2011) present the following<br />

definitions:<br />

1.) Electrode: electrical conductor submerged in electrolyte;<br />

2.) Electrolyte: liquid or gel that contains free ions, which<br />

can be decomposed by electrolysis;<br />

3.) Electrolysis: non-spontaneous process in which a direct<br />

current is used with the goal of obtaining a chemical<br />

reaction;<br />

4.) Anode: the electrode where reduction occurs (to which<br />

electrons move towards), known as the positive pole;<br />

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5.) Cathode: the electrode where oxidation occurs (from<br />

which electrons move away), also known as the negative<br />

pole.<br />

One of the manufacturers of zinc-air batteries<br />

(Energizer, 2004) states that electricity is obtained from the<br />

following electrolytic reactions:<br />

A.) Anode: Zn + 2OH ZnO + H2O + 2e<br />

B.) Cathode: O2 + 2H2O + 4e 4OH<br />

C.) Final: 2Zn + O2 2ZnO<br />

Reducing the terms gives the following reaction:<br />

Zn + O2 ZnO (1)<br />

Zinc (Zn) particles are mixed with an electrolyte,<br />

while water and oxygen in the air react to form hydroxides<br />

at the cathode, which migrate to form zincate (Zn(OH) 4<br />

2-<br />

). Electrons are released and flow to the cathode where<br />

reduction produces zincate oxide (ZnO), while water is<br />

returned to the system. Water and zinc hydroxide are<br />

recycled at the anode for the cathode, so that water<br />

serves as a catalyst for the reaction. A typical voltage<br />

resulting from this reaction is 1.4 V (Energizer, 2004). If<br />

stored at room temperature without removal of the seal<br />

to the air inlet, a zinc-air cell can store 95% of its capacity<br />

for one year, or 90% of its capacity for up to two years.<br />

Valente et al. (2007) state that the positive holes in<br />

batteries serve to reset the O 2<br />

battery, resulting in<br />

electric battery terminals (Formula 1). Zinc-air batteries<br />

are practical with good energy density, are stable and<br />

safe with a low voltage and current, and must be<br />

discarded after use (Wei et al., 2000). Zinc does not<br />

harm the environment, is easier to store than other<br />

materials (e.g., O 2<br />

), and can be processed in water-based<br />

electrochemical systems (Zhang, Bruce, and Zhang,<br />

2011).<br />

IEC60086-1:2006 and IEC60086-2:2006 are<br />

applicable to primary batteries and specify their physical<br />

dimensions as well as their test conditions and discharge<br />

performance requirements. The following terms are taken<br />

from the later standard:<br />

I.) Nominal voltage of primary battery: suitable<br />

approximate value of voltage used to identify the<br />

voltage of a primary battery;<br />

II.) End-point voltage: specified voltage of a battery at<br />

which the battery discharge is terminated;<br />

III.) Closed circuit voltage: voltage across the terminals of<br />

a battery when it is on discharge;<br />

IV.) Primary cell: one or more primary cells, including case,<br />

terminals, and marking;<br />

V.)<br />

Storage life: duration under specified conditions at the<br />

end of which a battery retains its ability to perform a<br />

specified service output;<br />

VI.) Terminals: conductive parts provided for the<br />

connection of a battery to external conductors;<br />

VII.) Application test: simulation of the actual use of a<br />

battery in a specific application;<br />

VIII.) Off-load voltage: voltage across the terminals of a<br />

battery when no current is flowing;<br />

IX.) Service output: service life, or capacity, or energy<br />

output of a battery under specified conditions of<br />

discharge;<br />

X.)<br />

Discharge: operation during which a battery delivers<br />

current to an external circuit;<br />

XI.) Leakage: unplanned escape of electrolyte, gas, or<br />

other material from a battery.<br />

Halliday and Resnick (1988) report the following<br />

definitions:<br />

a.) Voltage-electrical potential: difference between two<br />

poles, may be continuous voltage (one pole is always<br />

positive and the other always negative) or alternating<br />

current (the poles vary in their polarity);<br />

b.) Electrical current: orderly movement of electrons, which<br />

can be direct or alternating;<br />

c.) Electrical resistance: electrical component that hinders<br />

the passage of electric current, dissipating heat energy<br />

to the mean;<br />

d.) Load-end component: circuit that receives electrical<br />

current at its terminals;<br />

e.) Recharging the battery: the inverse process to the<br />

normal operation of a battery in which tension is<br />

applied at its terminals, to recombine the electrolyte to<br />

the battery so that it can continue to feed a load.<br />

Digital signal processors force the involuntary shut<br />

down of hearing aids when the supply voltage reaches 1.0<br />

V (SDT, 2007) by ensuring that the voltage is not sufficient<br />

to power the electronic circuit, preventing unpredictable<br />

behavior and avoiding insufficient amplification or<br />

occurrence of spurious signals with high levels of distortion.<br />

Kates (2008) states that a battery for a hearing aid should<br />

last for at least 50 hours.<br />

As we describe later, even battery manufacturers do<br />

not always make the technical data for their batteries<br />

available. Only one distributor of hearing aid batteries<br />

(Microbattery, 2013) provides an online table comparing<br />

the specifications of 10 brands, by presenting the data<br />

sheets from various manufacturers. However, this focuses<br />

on presenting information for marketing purposes in order<br />

to promote sales through their website.<br />

With this information in mind, it can be seen that<br />

it is difficult for otologists and audiologists to recommend<br />

the brand of hearing aid battery with the best performance<br />

to patients. Factors such as patient comparisons of battery<br />

brand durability, the risk of battery leaks and how to deal<br />

with them, procedures to use in the case of ingestion, how<br />

to dispose of hearing aid batteries, whether zinc-air<br />

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batteries can be recharged, and if rechargeable batteries<br />

fully replace zinc-air batteries have not been fully<br />

investigated. There are no publications in the literature<br />

addressing these basic concerns, although some<br />

information can be found in scientific publications and lay<br />

media.<br />

Thus, we attempted to address the above concerns<br />

and develop laboratory tests for the practical purpose of<br />

comparing the performance of 4 different sizes of 10<br />

brands of batteries for hearing aids that can be found in the<br />

Brazilian marketplace. We did not conduct a financial<br />

analysis between zinc-air batteries and rechargeable<br />

batteries. The primary objective of this research was to<br />

investigate the performance of 10 brands of hearing aid<br />

batteries under the same test conditions. The secondary<br />

objectives were to describe courses of action to be taken<br />

for battery leaks, accidental intake, and disposal.<br />

Battery identification<br />

Hearing aid pills are commonly marketed as batteries;<br />

therefore, we refer to pills, cells, and batteries as batteries<br />

in this work. Once the batteries were checked they were<br />

identified as ExtraPower (A), ClearCell (B), PowerOne (C),<br />

Renata (D), Energizer (E), Duracell (F), Rayovac (G),<br />

icellTech (H), Sony (I), or Panasonic (J). These identifications<br />

made it possible to avoid any confusion concerning the<br />

manufacturer and model, and were marked on the batteries<br />

with a knife. This marking system did not cause the<br />

exchange of heat with the batteries and thus did not<br />

change the electrolytes inside the batteries. Figure 1 shows<br />

a #675 battery by ExtraPower. Figure 2 shows a #675<br />

battery by ClearCell viewed under a microscope. Two<br />

packs were acquired from each of the manufacturers listed<br />

in Chart 1 so that we could retest any of the batteries as<br />

required (Figure 1 and 2).<br />

METHOD<br />

This study was performed in the Laboratory of<br />

Acoustics at the Department of Otorhinolaryngology<br />

between June <strong>17</strong>, 2011 and February 5, 2012.<br />

Battery acquisition<br />

Hearing aid batteries can be found in sizes #675,<br />

#13, #312, #10, and #5 (in descending size). In the<br />

Brazilian marketplace, it is hard to find hearing aids with<br />

batteries of size #5, so this size was not tested in the<br />

current study. Before battery acquisition, we performed<br />

a quick survey of experienced audiologists to determine<br />

which brands of hearing aid batteries were most used by<br />

them, and which brands patients recommended. With this<br />

information, manufacturers, dealers, and distributors were<br />

contacted by mail with a standard message, and,<br />

depending on the manufacturer, they sent us samples,<br />

data sheets, material safety data sheets, and marketing<br />

material. For example, Mazalab (ExtraPower) sent a free<br />

pack of batteries but no technical documents, while<br />

Ammon & Rizos (Renata) sent a battery pack and the<br />

respective data sheets. Although a survey of all the<br />

manufacturers was conducted, some did not provide data<br />

sheets for their products, namely ExtraPower, ClearCell,<br />

PowerOne (specifications only), and Sony. Possession of<br />

data sheets allowed observation of common technical<br />

standards and a direct comparison of the performance of<br />

batteries from several manufacturers. All the battery<br />

suppliers were located in the city of São Paulo. The six<br />

suppliers contacted are listed in Chart 1. The last three<br />

vendors listed in the chart donated their batteries, while<br />

the others sold them at retail value.<br />

Figure 1. ExtraPower #675 identification.<br />

Figure 2. ClearCell #675 identification.<br />

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Chart 1. General battery information.<br />

Supplier Manufacturer/ Model Size Identification/ Nominal Service Quantity/ Best<br />

brand designation voltage output pack before<br />

(V) (mAh)<br />

Digitall Panasonic PR675H 675 PR-675HEP/6C 1.4 605 6 10/2013<br />

PR13H 13 PH13HEP/6C 1.4 300 6 09/2013<br />

PR312 312 PR-312HEP/6C 1.4 <strong>17</strong>0 6 10/2013<br />

PR230H 10 PR-230HEP/6C 1.4 65 6 05/2013<br />

icellTech Platinum 675 PR44 1.4 630 6 10/2013<br />

Platinum 13 PR48 1.4 310 6 12/2013<br />

Platinum 312 PR41 1.4 180 6 05/2013<br />

Digital Sound 10 PR70 1.4 105 6 05/2012<br />

Joave Sony 675 675 675(PR44) 1.4 NI 6 02/2014<br />

13 13 13(PR48) 1.4 NI 6 10/2014<br />

312 312 312(PR41) 1.4 NI 6 10/2013<br />

10 10 10(PR70) 1.4 NI 6 10/2014<br />

Duracell Activair 675 PR44 1.4 600 4 08/2014<br />

Activair 13 PR48 1.4 290 4 11/2014<br />

Activair 312 PR41 1.4 150 4 12/2014<br />

Activair 10 PR70 1.4 90 4 02/2015<br />

Energizer AudioPRO 675 AC675-4AP 1.4 635 4 10/2012<br />

AudioPRO 13 AC13-4AP 1.4 280 4 10/2013<br />

AudioPRO 312 AC312-4AP 1.4 160 4 07/2012<br />

AudioPRO 10 AC10-4AP 1.4 91 4 01/2012<br />

PowerOne PowerOne 675 p675 1.4 650 6 07/2013<br />

PowerOne 13 p13 1.4 310 6 01/2014<br />

PowerOne 312 p312 1.4 180 6 01/2014<br />

PowerOne 10 p10 1.4 100 6 01/2014<br />

CTEA Rayovac Extra Advanced 675 675AE-6LD 1.45 NI 6 06/2014<br />

Extra Advanced 13 13AE 1.45 NI 6 08/2014<br />

Extra Advanced 312 312AE-6LD 1.45 NI 6 01/2014<br />

Extra Advanced 10 10AE-6LD 1.45 NI 6 10/2014<br />

SANCIEX ClearCell Premium 675 675 1.4 630 4 10/2013<br />

Premium 13 13 1.4 300 4 10/2013<br />

Premium 312 312 1.4 180 4 10/2013<br />

Premium 10 10 1.4 100 4 10/2013<br />

MazaLab ExtraPower ExtraPower 675 A675 1.4 630 6 12/2013<br />

ExtraPower 13 A13 1.4 300 6 07/2013<br />

ExtraPower 312 A312 1.4 180 6 07/2013<br />

ExtraPower 10 A10 1.4 100 6 07/2013<br />

Ammon & Rizos Renata Maratone+ 675 ZA675 1.4 650 6 05/2013<br />

Maratone+ 13 ZA13 1.4 310 6 11/2013<br />

Maratone+ 312 ZA312 1.4 180 6 03/2014<br />

Maratone+ 10 ZA10 1.4 100 6 05/2014<br />

NI - not informed.<br />

Tests details<br />

Tests were initialized 10 days after the receipt of<br />

all the batteries so that the effects of storage in the<br />

laboratory did not alter the performance of the batteries.<br />

With the possession of data sheets, it was observed that<br />

there is no standardization for performance evaluation.<br />

For example, Renata #675 follows standard IEC60086-2,<br />

which prescribes a discharge cycle of 12 hours in a load<br />

resistor of 619 Ω followed by a subsequent 12 hours of<br />

rest (for chemical recombination with subsequent partial<br />

recovery), with a temperature and humidity of 21 o C and<br />

50%, respectively. icellTech #675 prescribes a discharge<br />

cycle of 16 hours in a load resistor of 620 Ω followed by<br />

8 hours of rest, with an ambient temperature of 25 o C and<br />

a relative humidity of 50%, without mentioning any<br />

standard. Therefore, we chose to use fixed and high<br />

precision resistors (Chart 2) to simulate a fixed load in a<br />

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closed circuit voltage along with a prescribed discharge<br />

cycle of 12 hours followed by a subsequent 12 hour rest<br />

period. Both the values of the resistors, the charge and<br />

the discharge cycle, meet the requirements of IEC60086-<br />

2 (Table 7.4.2. in this standard) pertaining to each of the<br />

battery sizes.<br />

Chart 2. Resistor values for closed circuit voltage simulation.<br />

Battery size Load resistor (Ω) Precision (%)<br />

#675 620 1<br />

#13 1500 2<br />

#312 1500 2<br />

#10 3000 1<br />

Neither the temperature nor the humidity was<br />

controlled by an air conditioning system, on account of the<br />

fact that the laboratory where the tests were conducted<br />

does not have a precision system, only a split air conditioning<br />

system. However, both the temperature and the relative<br />

humidity were monitored by a Minipa hygrometer (MT241),<br />

and measurements were recorded randomly during testing<br />

(lowest 45%, highest 74%). We used a calibrated Minipa<br />

multimeter (ET-2042C) to measure voltages.<br />

All battery seals were removed from zinc-air batteries<br />

and the batteries were then left for 10 minutes for activation<br />

(Renata ref. ZA675 Maratone+ Rev. 3/January 2009),<br />

although Energizer (2004) recommends 30 seconds for the<br />

same procedure.<br />

It was necessary to assemble a test jig (Figure 3),<br />

i.e., a dedicated device for assisting in the evaluation of<br />

battery performance. For the construction of the test jig,<br />

only discrete components such as resistors, relays, switches,<br />

fuses, and connectors were used, and the logical drive was<br />

handled by an EcoGold programmable timer (EG-TMR009;<br />

Figure 4).<br />

Figure 3. Test jig, top view.<br />

The general scheme of operation of the jig can be<br />

seen in Figure 5.<br />

A closed circuit composed of a resistor and a relay<br />

(in which electrical contacts opened or closed its<br />

corresponding circuit) was used to test each battery<br />

individually. This design met the specifications of<br />

IEC60086-2 fixed-charge. Thus, the battery (P1) fed the<br />

load resistor (R1; as shown in Chart 1) by contact S1 (K1<br />

relay). This configuration was repeated 24 times. A<br />

general key (S) enabled or did not enable the circuit,<br />

while a fuse (F) protected against test jig overcurrent. A<br />

direct current supplier (P; AC to DC converter) fed the<br />

relay coils, according to the schedule set in the<br />

programmable timer (T). The timer was powered by<br />

mains 110 V conventional R.<br />

Through the test jig, it was possible to test two<br />

batteries of each brand simultaneously. Two batteries of<br />

each brand were used in simultaneous testing so that the<br />

worst of the two values could be excluded; thus, only the<br />

best performance for each of the sizes and brands of each<br />

battery was considered. In this way, up to 44 batteries were<br />

tested simultaneously in the test jig.<br />

Figure 4. Programmable timer, top view.<br />

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Measurements were conducted immediately, following<br />

the sequence A, B, C, (...) until the last battery. The next<br />

day, measurements started with the last battery and followed<br />

the sequence backwards to battery A. On holidays, Saturdays,<br />

and Sundays the building facilities were closed, thus a<br />

different procedure was required. The night before a<br />

holiday, Saturday, or Sunday, 15 minutes was added to the<br />

programmable timer, so as to allow any additional battery<br />

discharge before measurements. Therefore, we sought to<br />

offset the effects of partial recombination charge, which<br />

could have masked the last readings of voltage, especially<br />

at the beginning of testing, when the batteries had a higher<br />

capacity for recombination of partial charge.<br />

All batteries were tested under this scheme. A<br />

flowchart detailing the logic employed in the test is shown<br />

in the Annex.<br />

To record images, we used a Leica binocular<br />

microscope (EZ4) and a Samsung digital camera (SL30).<br />

RESULTS<br />

The results are graphs 1 to 4 and Table 1.<br />

Figure 5. Simplified schematics implemented in the test jig.<br />

To ensure that each circuit was functioning individually,<br />

the multimeter was used to verify if there was voltage across<br />

each of the resistors, which indicated that the circuit was<br />

functioning correctly. Twice daily voltage measurements<br />

were performed in a random order at each of the resistors.<br />

After measuring the initial voltages of the batteries<br />

without load (open circuit voltage), they were then<br />

subjected to load (closed circuit). These cycles were<br />

alternated in accordance with a prescribed logic, to assess<br />

how many hours a battery would last until its final voltage<br />

of 1.1 V. This was followed by four more measurements, in<br />

order to ensure that once a battery had reached this value,<br />

it was unable to supply voltage (end-point voltage). The<br />

value of 1.1 V was used as the cutoff voltage because it was<br />

higher than the amount proposed by SDT (2007) with<br />

some safety margin and, in general, it is observed that<br />

values below 1.1 V can cause unpredictable behavior of<br />

hearing aids. In addition, the manufacturers’ data sheets<br />

showed that at around this value, zinc-air batteries have<br />

virtually no more capacity for power supply.<br />

Accordingly, the programmable timer was set to<br />

energize the test jig at 10:05 am following a discharge cycle<br />

of 12 hours, which peaked at 8:05 am on the next morning.<br />

Graphic 1. Discharge curve of zinc-air size #675.<br />

Graphic 2. Discharge curve of zinc-air size #13.<br />

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Annex. Logic implemented on tests<br />

Graphic 3. Discharge curve of zinc-air size #312.<br />

Graphic 4. Discharge curve of zinc-air size #10.<br />

Table 1. Zinc-air battery values (average and standard<br />

deviation).<br />

Size Average service (hours) Nominal voltage (volts)<br />

#675 (270 ± 45) (1.357 ± 0.039)<br />

#13 (266 ± 26) (1.376 ± 0.057)<br />

#312 (199 ± 32) (1.370 ± 0.068)<br />

#10 (245 ± 15) (1.339 ± 0.067)<br />

DISCUSSION<br />

The necessity to use a standard for evaluating the<br />

performance of hearing aid batteries may be questioned,<br />

since such evaluation can be performed by experienced<br />

patients or driven by engineers. For example, each patient<br />

has a need for selective amplification, and amplification is<br />

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related to battery consumption in the following ratio: the<br />

higher the amplification, the greater the battery drain. In<br />

general, the batteries tested in the current study had the<br />

capacity to supply power for up to 270 hours (Table 1).<br />

This means that patients would need to continue to test<br />

batteries under the same conditions for about 22 days (270<br />

hours divided by 12 hours/day). It would be a hindrance to<br />

the patient to maintain rigorous battery use at the same<br />

volume for 22 consecutive days. Furthermore, this procedure<br />

would have to be repeated for all brands in the four sizes<br />

of batteries available. Battery drain varies greatly with<br />

acoustic feedback. When a hearing aid mold is not well<br />

coupled to the ear (BTE design hearing aid cases), this is<br />

enough to produce feedback. Tests conducted in our<br />

laboratory indicate that when feedback occurs, hearing aids<br />

have a current drain (4.34 mA, Figure 7) several times<br />

higher than a hearing aid without feedback (0.94 mA,<br />

Figure 6). Thus, while tests with patients may directly<br />

transcribe their views, assessment of hearing aid batteries<br />

demands specialized and sequentially standardized testing,<br />

which is only possible using application tests in a laboratory.<br />

The objectives of the present study included testing<br />

batteries within the conditions prescribed by IEC60086-1,<br />

IEC60086-2, and IEC60086-3; however, this was not always<br />

possible since the laboratory where this study was performed<br />

is not one of a certifying body, but one of a battery<br />

consumer. For example, the above referenced IEC standards<br />

require testing of nine batteries of the same brand and<br />

model (this study tested two batteries of each brand and<br />

model), and combine the measurement of physical battery<br />

size with electrical testing with fixed load (performed in<br />

this study) and load with standard and high pulse drain (not<br />

performed in this study). Additionally, both the temperature<br />

and the relative humidity should vary only under restricted<br />

margins, which can be seen in Tables 4 and 7 of IEC60086-<br />

1. In this regard, the storage temperature of batteries must<br />

be between +10 o C and +25 o C (50 o F and 77 o F, respectively),<br />

and should never exceed +30 o C (86 o F), which makes<br />

storage at low temperatures (-10 o C up to +10 o C or 14 o F up<br />

to 50 o F) ideal, either for testing purposes or for marketing<br />

purposes.<br />

However, Annex G of IEC60086-1 defines that any<br />

battery consumer can establish a standard methodology for<br />

measuring the performance of batteries (SMMP) that meets<br />

the following criteria:<br />

a) The test methods should be defined in such a way that<br />

the test results correspond as closely as possible to the<br />

performance results as experienced by consumers<br />

when using the product in practice;<br />

b) It is essential that the test methods are objective and<br />

give meaningful and reproducible results;<br />

c) Details of the test methods should be defined with a<br />

view to optimum usefulness to the consumer, taking<br />

Figure 6. Hearing aid curves without feedback.<br />

Figure 7. Hearing aid curves with feedback.<br />

into account the ratio between the value of the product<br />

and the expenses involved in performing the tests;<br />

d) Where use has to be made of accelerated test procedures,<br />

or of methods that have only an indirect relationship to<br />

the practical use of the product, the technical committee<br />

should provide the necessary guidance for correct<br />

interpretation of test results in relation to normal use of<br />

the product.<br />

The present study meets the above criteria, so its<br />

results allow a performance analysis comparison between<br />

batteries of various brands.<br />

It was observed that battery manufacturers do not<br />

always provide data sheets for their products, making it<br />

difficult to compare the performance of various battery<br />

brands. The data sheets that were provided did not follow<br />

a specific standard, which again made it difficult to compa-<br />

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re the results obtained in this experiment with the<br />

manufacturers’ data. Among the restrictions for comparison<br />

of performance, it was found that the documentation<br />

provided by PowerOne transcribed only a few numerical<br />

specifications (voltage, typical energy, and capacity), and<br />

thus abstained from describing the dynamic behavior of the<br />

battery. This can be described by graphs, in a similar way<br />

to the data sheets provided by Renata, Energizer, Duracell,<br />

Rayovac, and icellTech. The Panasonic data sheet only<br />

showed dynamic curves of high drain, resulting in a<br />

disturbing and misguided image of low battery performance<br />

for this manufacturer.<br />

Comparing the manufacturers’ data sheets with the<br />

curves obtained in the current study highlighted many<br />

discrepancies. To this end, we inserted a line parallel to the<br />

abscissa axis until it crossed the voltage of 1.1 V both in the<br />

graphs provided in data sheets and the graphs obtained<br />

using the procedure described in this study. Thus, the<br />

discharge time was defined as the time (in hours) that the<br />

batteries provided voltage above 1.1 V. The following<br />

table shows the data for each battery from each of the<br />

manufacturers.<br />

From Table 2 it can be seen that for batteries of size<br />

#675, there was large variation between the values listed<br />

in the data sheets (lowest value = 110 hours, highest value<br />

= 560 hours) compared to the values measured in this<br />

study (lowest value = 240 hours, highest value = 372<br />

hours). While the average service measured in this study<br />

(Table 1) was (270 ± 45) hours, the average service from<br />

the data sheets (Table 2) was (290 ± 161) hours.<br />

Table 2 also shows that batteries of size #13 showed<br />

wide variation in their data sheets (lowest value = 140<br />

hours, highest value = 440 hours) when compared with the<br />

values obtained in this study (lowest value = 240 hours,<br />

highest value = 300 hours). Table 1 indicates that the<br />

average service measured was (266 ± 26) hours, while the<br />

data sheets (Table 2) showed a final measurement of (306<br />

± 107) hours.<br />

Regarding batteries of size #312, there was great<br />

variability among the values of the data sheets (lowest<br />

value = 140 hours, highest value = 360 hours) compared to<br />

the values obtained in this study (lowest value = 156<br />

hours, highest value = 276 hours). The average service<br />

measured was (199 ± 32) hours, whereas the same<br />

parameter from the data sheets (Table 2) was (204 ± 88)<br />

hours.<br />

Finally, the data sheets for batteries of size #10<br />

showed considerable variation (lowest value = 130 hours,<br />

highest value = 360 hours) compared to the values<br />

obtained by measurements made in this study (lowest<br />

Table 2. Average service time taken from the manufacturers’<br />

data versus the results of the current study.<br />

Manufacturer Size Average Average Difference<br />

service 1 service 2 (%)<br />

(hours) (hours)<br />

Renata #675 560 240 57<br />

#13 440 300 32<br />

#312 360 204 43<br />

#10 360 240 33<br />

Energizer #675 290 264 9<br />

#13 310 240 23<br />

#312 180 156 13<br />

#10 190 252 -33<br />

Duracell #675 280 372 -33<br />

#13 310 286 8<br />

#312 <strong>17</strong>0 276 -62<br />

#10 180 252 -40<br />

Rayovac #675 110 252 -129<br />

#13 140 276 -97<br />

#312 140 204 -46<br />

#10 130 276 -112<br />

icellTech #675 290 240 <strong>17</strong><br />

#13 330 240 27<br />

#312 <strong>17</strong>0 204 -20<br />

#10 208 252 -21<br />

Notes:<br />

1.) Data from manufacturers’ data sheets;<br />

2.) Data from the current study.<br />

value = 240 hours, highest value = 276 hours). The data in<br />

Table 1 show that the average service obtained in this<br />

study was (245 ± 15) hours, whereas the data sheets (Table<br />

2) indicated a value of (214 ± 87) hours for the same<br />

parameter.<br />

Taken together, the information provided above<br />

highlights the fact that there was a large variability in the<br />

values presented in the manufacturers’ data sheets, while<br />

laboratory test results reflected a lower variability, which<br />

confirms the reliability of tests performed in the laboratory.<br />

Asymmetric information was provided in data sheets, e.g.,<br />

the data sheet of one manufacturer (icellTech #675) had<br />

an average service of 290 hours and that for a battery of the<br />

same size but from another manufacturer (Renata) had an<br />

average service of 560 hours (93% variation). It is not clear<br />

what the technological aspects were that enabled this<br />

intriguing variation in performance from one manufacturer<br />

to the other, since it is assumed that existing technology<br />

was not manipulated in a significantly different way. For<br />

example, when these batteries were subjected to the same<br />

dynamic tests, both had a discharge time of 240 hours. In<br />

addition, with regards to batteries of size #675, it is difficult<br />

to explain how Rayovac battery data sheets showed an<br />

average service of only 110 hours, as compared to 290<br />

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hours and 560 hours in the data sheets of Renata and<br />

icellTech, respectively. In dynamic testing, Rayovac<br />

batteries had an average service of 252 hours, which was<br />

close to the values registered for Renata and icellTech<br />

batteries (240 hours). Similar results were obtained with<br />

other batteries.<br />

In a similar way, the data sheet for size #13 Rayovac<br />

batteries reported an average service of 140 hours, while<br />

Renata’s data sheet stated a service of 440 hours, which is a<br />

variation of more than 200%. When this was measured<br />

dynamically, the values changed by 25% (240 hours and<br />

300 hours, respectively). Similar results were observed with<br />

respect to batteries of size #312, since the Renata data<br />

sheets reported an average service of 380 hours, Rayovac’s<br />

stated a service of 140 hours, and icellTech’s stated a service<br />

of about <strong>17</strong>0 hours, whereas the dynamic values obtained<br />

in this study indicated a service time of 204 hours. A similar<br />

result was also found with respect to size #10 batteries: the<br />

data sheet from Renata stated an average service of 370<br />

hours, whereas those from Energizer and Duracell stated 190<br />

hours and 200 hours, respectively. Laboratory tests indicated<br />

the values of 240, 252, and 252 hours, respectively.<br />

The American brands Energizer, Duracell, and<br />

Rayovac have data sheets referencing both IEC60086-2<br />

and American standards (ANSI-7003ZD battery #675, ANSI-<br />

7000ZD battery #13, ANSI-7002ZD battery #312, and<br />

ANSI-7005ZD battery #0). The American and European<br />

standards are not identical, so there are variations in the<br />

presentation of results, making it difficult to compare the<br />

performance of American manufacturers with that of<br />

European manufacturers. Ideally, the adoption of one<br />

pattern should be applied in Brazil. Manufacturers should<br />

also provide data sheets in Portuguese, which would lead<br />

to a better understanding of the product by the reader. This<br />

would also make the reader feel valued as a result of their<br />

native language being included in the product information.<br />

The results for the size #675 batteries can be divided<br />

into three distinct groups: a group composed of six brands<br />

with similar behavior, an intermediate group consisting of<br />

three brands with a slight descent in performance, and a<br />

final group of one brand with the best performance. Thus,<br />

the worst batteries in terms of performance were ClearCell,<br />

ExtraPower, icellTech, Sony, Rayovac and Renata (average<br />

of 250 hours), the intermediate group comprised Energizer<br />

(264 hours), PowerOne (300 hours), and Panasonic (312<br />

hours), and best performance Duracell (372 hours).<br />

Battery performance for size #13 batteries can also<br />

be divided into three groups: a group composed of four<br />

brands with similar performance, a second group comprising<br />

also four brands with a slight descent in performance, and<br />

a third group containing the top two brands. ClearCell,<br />

ExtraPower, icellTech, and Energizer showed the worst<br />

performance (240 hours), followed by Panasonic, Duracell,<br />

, PowerOne, and Rayovac (264, 276, 276, and 276 hours,<br />

respectively), with Renata (300 hours) and Sony (312<br />

hours) showing the best performance.<br />

The results for batteries of size #312 can be divided<br />

into four groups: a first group containing the worst<br />

performers, a second group with two brands whose results<br />

were slightly higher in relation to the previous group, a<br />

third group consisting of five brands with similar results, and<br />

a fourth consisting of one brand . Energizer (156 hours) and<br />

ExtraPower (168 hours) make up the first group, ClearCell<br />

(180 hours) and PowerOne (192 hours) follow in the<br />

second group; icellTech, Renata, Sony, Panasonic, and<br />

Roayovac (204 hours) comprise the third group, and<br />

Duracell (276 hours) in the fourth group.<br />

Three groups can also be used to categorize the<br />

behavior of #10 batteries. The first group is constituted by<br />

the four worst performers, followed by a second group with<br />

slight higher performance, and finally, the brands with the<br />

best performance. ExtraPower and ClearCell (228 hours),<br />

PowerOne and Renata (240 hours) constitutes the first<br />

group; the second group consists of Duracell, Sony, icellTech,<br />

and Energizer ( 252 hours); Panasonic (264 hours) and<br />

Rayovac (276 hours) are the brands in the last group.<br />

Zinc-air batteries are usually referred as mercury<br />

free, when, in fact, some have mercury in their constitution.<br />

For example, icellTech states that they use less than 25 mg<br />

of mercury per battery (icellTech, 2004) while Duracell<br />

(2008) reports that their batteries satisfy the limit of 0.1<br />

mg/m 3 . Zhang, Bruce, and Zhang (2011) state that despite<br />

the benefits of reduced mercury in zinc-air batteries, there<br />

are still some disadvantages such as limitation of power<br />

(low voltage and current), evaporation of the electrolyte<br />

(failure due to the entry of air into the battery), adverse<br />

reactions arising from the presence of other gases in the<br />

environment (e.g., the entry of carbon dioxide can produce<br />

solid carbide), and production of solid elements (difficult<br />

and costly to dispose of). These authors suggest that<br />

lithium-air technology may replace zinc-air batteries in the<br />

near future.<br />

Cochlear implants also require batteries, for two<br />

functions: operation of the transmission electronic circuit<br />

and voltage as well as operation of the modulated radio<br />

frequency signal from the antenna external to the implanted<br />

module (Clark, 2008; Sit and Sarpeshkar, 2008; Bhoir and<br />

Panse, 2009); this leads to high current consumption<br />

(Wilson, 2004; Zeng, Rebscher, and Harrison, 2008). It is<br />

therefore recommended to test a larger number of<br />

manufacturers and models of batteries within the hearing<br />

aid conditions of any specific technical standard. Further<br />

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studies could incorporate testing batteries for cochlear<br />

implants. In this case, it would be feasible to perform a<br />

financial analysis of zinc-air batteries and rechargeable<br />

ones, given that the latter are very expensive compared to<br />

the former, but may be used several times.<br />

Rechargeable batteries<br />

Solar Ear distributes rechargeable batteries for hearing<br />

aids in Brazil. They sent us eight batteries in sizes #675 and<br />

#13 to test, plus two solar chargers (Figure 8). These solar<br />

charger hearing aid batteries are not charged directly by<br />

solar energy, since solar energy is only used to charge the<br />

two #AA batteries inside the charger; instead, the #AA<br />

batteries function as accumulators of electricity for charging<br />

the hearing aid batteries. This process is possible because<br />

#AA batteries have a higher load capacity than hearing aid<br />

batteries, allowing a controlled discharge of current from<br />

#AA batteries to hearing aid batteries. This also occurs<br />

when there is electricity in the internal #AA batteries<br />

controlled by an electronic circuit located in the charger.<br />

As stated in the solar charger user’s manual (in<br />

English, without references) it is necessary to charge the<br />

internal batteries before charging hearing aid batteries.<br />

According to this manual, the charging process can be<br />

performed in two ways: solar charging (by leaving the<br />

charger exposed to sunlight or artificial light) or through an<br />

external battery charger. We decided to remove the #AA<br />

batteries from the solar charger and left it for six hours to<br />

charge via an external charger (Duracell, CEF14N model -<br />

not supplied), as shown in Figure 9. We did this because<br />

if the batteries were recharged by the #AA solar charger,<br />

this would require 20 hours of exposure to the sun or a light<br />

source. Additionally, our laboratory does not have a window<br />

that receives direct sunlight, rainfall may have disrupted<br />

charging, or the solar charger may even have fallen and<br />

been damaged if it was not positioned in a safe place. In<br />

urban locations, it is more practical to utilize the convenience<br />

of residential electricity, a USB connector (universal serial<br />

bus: standard connection to a computer and its peripherals<br />

that can rely on a DC power supply), or even power from<br />

a car than actual solar energy. This is a result of the difficulty<br />

in obtaining direct sunlight for the solar charger, caused by<br />

buildings that obscure the availability of natural light in the<br />

environment, not to mention loading restrictions on cloudy<br />

or rainy days. Therefore, removal of the #AA batteries and<br />

external charging was the best option.<br />

Thus, the #AA batteries were charged and put back<br />

in the solar charger. Then the process of charging the #675<br />

and #13 batteries was started, by inserting them into the<br />

connectors on the solar charger and waiting until the green<br />

LED solar charger light went out. The charging time lasted<br />

Figure 8. Solar chargers.<br />

Figure 9. Rechargeable #AA batteries.<br />

8 hours for #675 batteries and six hours for #13 batteries,<br />

as stated in the user’s manual. During the tests, Solar Ear<br />

provided us with a new version of the solar charger which<br />

had an input for an external power supply (Figure 8, right),<br />

but we preferred to continue to use the first solar charger<br />

supplied (Figure 8, left).<br />

According to IEC60086, zinc-air batteries cannot be<br />

compared directly with rechargeable batteries, because<br />

the former are said to be primary cells while the later are<br />

said to secondary batteries. However, such comparison is<br />

inevitable in terms of the average service given, which is<br />

one of the parameters considered when making the<br />

decision regarding use of one battery or the other. Thus,<br />

the rechargeable batteries were tested with the same<br />

procedures described for zinc-air batteries. In total, eight<br />

batteries of both sizes were subjected to tests, the results<br />

of which are represented in two formats: as absolute values<br />

(Table 3) and as a graphic version (Graphic 5). Solar Ear<br />

batteries have proper identification on the anode (Ni-MH<br />

cell HL40H and Ni-MH for #675 and #13, respectively), so<br />

there was no need for battery identification.<br />

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above 1.1 V as “the battery may be underutilized resulting<br />

in insufficient use of the available capacity.”<br />

The charge capacity of the rechargeable batteries<br />

was 50 and 18 mAh for the #13 and #675 sizes, respectively.<br />

By way of comparison, the #675 Duracell battery had a<br />

load capacity of 600 mAh, which is 12 times that of the<br />

same size Solar Ear battery, whereas the #13 Duracell<br />

battery had a load capacity of 290 mAh, which is 16 times<br />

the capacity of the same size Solar Ear battery.<br />

Graphic 5. Discharge curves of Solar Ear rechargeable<br />

batteries in sizes #675 and #13<br />

Table 3. Rechargeable battery values (average and standard<br />

deviation).<br />

Size Average service (hours) Nominal voltage (volts)<br />

#675 (58 ± 5) (1.229 ± 0.133)<br />

#13 (36 ± 10) (1.240 ± 0.115)<br />

Since the data sheets of the rechargeable batteries<br />

did not mention test conditions or related regulations, we<br />

cannot comment with regards to agreement or<br />

disagreement with their respective standards. However,<br />

judging by their capacity (50 mAh and 18 mAh for #675<br />

and #13 batteries, respectively), a higher average service<br />

was noted for zinc-air batteries than for rechargeable<br />

batteries. This observation is reaffirmed when the results<br />

of Tables 1 and 3 are compared. From these tables, it can<br />

be seen that #675 zinc-air batteries had an average<br />

service greater than five times that of the same size<br />

rechargeable batteries. A similar result was found for #13<br />

zinc-air and rechargeable batteries (average service seven<br />

times greater). The average values of all voltage<br />

measurements of zinc-air batteries were superior to those<br />

of the rechargeable batteries due to the higher load<br />

capacity of zinc-air batteries.<br />

The Solar Ear battery data sheet (Ni-MH cell button<br />

technical data) recommends that the cutoff of its products<br />

is 1.0 V, which results in a small variation (<strong>17</strong>%) of tension<br />

relative to their nominal voltage (1.2 V) as compared with<br />

the same parameter of zinc-air batteries for the same<br />

value of shear stress (29%). The same Solar Ear battery<br />

data sheet emphasizes that the cutoff for hearing aids is<br />

Varta (Varta Microbattery GmbH, Ellwangen,<br />

Germany) produces zinc-air batteries under the brand<br />

PowerOne (tested in the present study) as well as the<br />

rechargeable ACCU plus series in sizes #675, #13, #312,<br />

and #10 (Varta Material Safety Data Sheet MSDS 2,001,002,<br />

9/28/2009 Edition). These have a charger in the format of<br />

a pen (pencharger) that works similarly to the Solar Ear<br />

model, i.e., it has two #AAA batteries inside that carry load<br />

to hearing aid batteries housed in the charger. Varta also<br />

produces a pocket charger (pocketcharger) for #13,<br />

#312, and #10 batteries, the energy of which is stored in<br />

a rechargeable internal lithium charger. Exclusively for<br />

#675 batteries, Varta has a unique charger model<br />

(PowerOne 675 charger). All rechargeable batteries from<br />

Varta have a nominal voltage of 1.2 V that, according to<br />

IEC61951-2, provides the following average service values:<br />

74, 23, 31, and 12 mAh for #675, #13, #312, and #10<br />

batteries, respectively. We could not find a distributor of<br />

Varta rechargeable batteries in Brazil, so these batteries<br />

were not tested in the present study.<br />

In total, 180 zinc-air batteries were acquired (Chart<br />

1) and 96 were tested according to the methodology<br />

described. In addition, 16 rechargeable batteries were<br />

tested. All the batteries were segregated and will be<br />

returned to the manufacturers for disposal.<br />

Leakage<br />

Energizer (2011) states that zinc-air batteries should<br />

neither be recharged or wired in an inverted way nor<br />

placed in a short circuit because such events can cause<br />

leakage or explosion, and in extreme cases there may be<br />

injury to the operator. To prevent leakage, Panasonic<br />

(2010) stresses that their zinc-air batteries should not be<br />

deformed or mixed or soldered with other batteries.<br />

icellTech (2004) adds that its zinc-air batteries cannot be<br />

dismantled or placed in a fire as there is a risk of leakage<br />

or even explosion. The latter manufacturer stresses that<br />

the content of zinc-air batteries can cause irritation or<br />

burns to the skin; if this occurs, contamination must be<br />

removed with a cloth moistened with soap and water. In<br />

the case of contamination of the eyes, they must be<br />

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washed with water for a minimum of 10 minutes and then<br />

specialist help should be sought immediately. Duracell<br />

(2008) states that in the event of leakage, their zinc-air<br />

batteries would release caustic potassium hydroxide,<br />

reaching a total volume of 2.5 mL. If there is leakage of<br />

the hearing aid battery, the patient should seek assistance<br />

directly from the manufacturer of the device, as the use<br />

of cleaner can cause stains or irreversible damage to<br />

hearing aids. Leaks inside hearing aids invariably and<br />

permanently damage electronic components (digital signal<br />

processor, microphone, and receiver) and electrics (battery<br />

contacts, switches, and wiring). This can be avoided if<br />

only batteries from a good supplier within their validity<br />

are used and preventive maintenance of hearing aids is<br />

performed within the time recommended by the<br />

manufacturer.<br />

Accidental intake<br />

icellTech (2004) explains that continuous exposure<br />

to its zinc-air batteries does not cause any harmful effects<br />

on health, but in case of ingestion, the advice of a doctor<br />

should be sought immediately. However, Duracell (2008)<br />

warns that if accidental intake occurs, batteries must be<br />

recovered immediately as they can cause burning or<br />

perforation of the esophagus; it is not recommended to<br />

induce vomiting.<br />

Litovitz et al. (2010) describe a longitudinal study of<br />

8,648 cases of battery ingestion. When lodged in the<br />

esophagus, it was possible for batteries to cause severe<br />

damage simply within two hours of ingestion. Damage<br />

could progress to esophageal perforation,<br />

tracheoesophageal fistula appearance, vocal cord paralysis,<br />

and even death (13 deaths were recorded). Of all the cases<br />

of ingestion, 36% were reported to involve hearing aid<br />

batteries. In a previous study with 2,382 cases, Litovitz and<br />

Schmitz (1992) reported 952 cases (32%) of the accidental<br />

intake of hearing aid batteries.<br />

Proper disposal<br />

Brazilian law covers some aspects of battery disposal.<br />

From an institutional perspective, the Brazilian Association<br />

of the Electrical and Electronics Industry (Associação Brasileira<br />

da Indústria Eletro e Eletrônica - ABINEE) maintains<br />

a policy of disposal of solid waste based on the National<br />

Environmental Council (Conselho Nacional do Meio Ambiente<br />

- CONAMA) Resolution 401/2008 called the Reverse<br />

Logistics Program for Household Batteries, which has<br />

deployed systems and logistics for disposal after the end of<br />

life for ordinary batteries, zinc-manganese alkaline batteries,<br />

rechargeable batteries, and portable batteries. Zinc-air<br />

batteries are a subclass of zinc-manganese batteries. This<br />

policy also states the ABINEE goal in Resolution 257/99 of<br />

CONAMA, which reads:<br />

“Article 1 o : Batteries in their compositions containing<br />

lead, cadmium, and mercury and its compounds, necessary<br />

for the operation of any type of equipment, vehicles, or<br />

systems, mobile or fixed, as well as electric and electronic<br />

products that contain batteries integrated into their structure<br />

so that they are not replaceable after energy depletion, will<br />

be delivered to users by establishments that sell or network<br />

authorized services by the respective industries, to be<br />

transferred to manufacturers or importers, so that they<br />

adopt, directly or through third parties, the procedures for<br />

environmentally sound reuse, recycling, treatment, or<br />

disposal.”<br />

From the state perspective, the Department of the<br />

Environment of São Paulo defined SMA Resolution 38/<br />

2011, among other guiding “principles of vision and<br />

systemic post-consumer responsibility,” which implies the<br />

direct responsibility of manufacturers, importers, and<br />

distributors for their waste, even after the consumption of<br />

products, without defining the processes for allocation. A<br />

previous law (Law No. 12.300/2006 State, Article 53) has<br />

already highlighted the fact that:<br />

“Manufacturers, importers, or distributors of products<br />

require or may require special systems for packaging,<br />

storage, collection, transportation, treatment, or disposal, in<br />

order to avoid damage to the environment and public<br />

health, even after consuming their waste from these items,<br />

and are responsible for servicing requirements set by the<br />

environmental agency.”<br />

From the federal perspective, the CONAMA has a<br />

National Policy on Solid Waste instituted by Law No.<br />

12.305/10, which provides for the creation of systems of<br />

reverse logistics for collection and disposal of electronic<br />

products, lubricating oils, fluorescent lamps, packaging in<br />

general, and drugs. However, the Ministry itself (Brazil,<br />

2013) warns that “other products, however, as there are<br />

already regulations in place in this regard, such as those<br />

relating to pesticides and batteries, and tires and oil,<br />

working groups were not created to discuss reverse logistics<br />

in these chains.” However, this legislation appears to lack<br />

a comprehensive system that can handle marketing,<br />

disposal, collection, allocation, monitoring, evaluation, and<br />

feedback.<br />

Within this setting, Panasonic (2010) states that if its<br />

zinc-air batteries are thrown to the ground, they will leak<br />

electrolyte; they have not, however, assessed the possible<br />

resulting environmental damage. Energizer (2011) merely<br />

report that “disposal must comply with federal, state, or<br />

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Performance analysis of ten brands of batteries for hearing aids.<br />

Penteado et al.<br />

local law” and “technologies such as incineration or landfill<br />

should be considered.”<br />

CONCLUSION<br />

It was possible to investigate the performance of<br />

ten brands of hearing aid batteries, in addition to describing<br />

the procedures recommended for leakage, accidental<br />

intake, and disposal.<br />

REFERENCES<br />

1. Bloom S. Today’s hearing aid batteries pack more power<br />

into tinier packages. Hearing J. 2003;56(7):<strong>17</strong>-24.<br />

2. Bocchi N, Ferracin LC, Biaggio SR. Pilhas e baterias:<br />

funcionamento e impacto ambiental. Química Nova na<br />

Escola. 2000;11:3-9.<br />

3. Brasil. 2013. [cited 2013 Feb 13]. Available from: http:/<br />

/www.mma.gov.br.<br />

4. Cudahy E, Levitt H. Digital hearing aids: a historical<br />

perspective. In: Sandlin, RE. Understanding digitally<br />

programmable hearing aids. San Diego: Allyn and Bacon;<br />

1994.<br />

5. Duracell. Material Safety data Sheet. Bethel (US): Duracell;<br />

2008 5 p. Report No.: GMEL#2019-5.<br />

6. Energizer. Application Manual Zinc-Air (Zn/O2). 4 p.<br />

Detroit (US): Energizer; 2004.<br />

7. Energizer. Product Safety Datasheet. Detroit (US):<br />

Energizer; 2011 4 p.<br />

8. Halliday D, Resnick R. Física básica Rio de Janeiro. Livros<br />

Técnicos e Científicos; 1988. p. 95-125.<br />

9. Harvey D. Hearing aids. 1st ed. Sydney (Australia):<br />

Boomerang Press; 2000.<br />

10. icellTech. Material Safety Data Sheet. Seoul (Korea):<br />

icellTech; 2004 3p. Report No.: QP0502-4.<br />

11. Kates JM. Digital hearing aids. Plural Publishing: San Diego;<br />

2008.<br />

12. Knutsen JE. Power Supplies for Hearing Aids. Br J Audiol.<br />

1982;16(3):189-91.<br />

13. Litovitz T, Schmitz BF. Ingestion of Cylindrical and Button<br />

Batteries: An Analysis of 2382 Cases. Pediatrics.<br />

1992;89(4):747-57.<br />

14. Litovitz T, Whitaker N, Clark L. Preventing Battery<br />

Ingestions: An Analysis of 8648 Cases. Pediatrics.<br />

2011;125(6):1<strong>17</strong>8-83.<br />

15. Lybarger SF. A historical overview. In: Sandlin RE, editor.<br />

Handbook of hearing amplification. San Diego: Singular<br />

Publishing Group; 1988. p. 01-29.<br />

16. Microbattery. 2013. [cited 2013 Jan 22]. Available from:<br />

http://www.microbattery.com.<br />

<strong>17</strong>. Panasonic. Product Safety Datasheet. Osaka (Japan):<br />

Panasonic; 2010 5p.<br />

18. Pinkwart K, Tuebke J. Thermodynamics and mechanistics.<br />

In: Daniel C, Besenhard JO, editors. Handbook of battery<br />

materials. Weinhiem (Germany): Wiley-VCH Verlag & Co.<br />

KGaA; 2011. p. 03-26.<br />

19. SDT - Sound Design Technologies. Using DSP hybrids<br />

in high power applications. Burlington (Canada); 2007 12<br />

p. Report No 24561-2.<br />

20. Sparkes C, Lacey NK. A study of mercuric oxide and<br />

zinc-air battery life in hearing aids. J Laryngol Otolol.<br />

1987;111(9):814-9.<br />

21. Valente M, Cadieux JH, Flowers L, Newman JG, Scherer<br />

J, Gephart G. Differences in Rust in Hearing Aid Batteries<br />

across Four Manufacturers, Four Battery Sizes, and Five<br />

Durations of Exposure. J Am Acad Audiolol.<br />

2007;18(10)846-62.<br />

22. Wei Z, Huang W, Zhang S, Tan J. Carbon-based air<br />

electrodes carrying MnO2 in zinc–air batteries. Journal of<br />

Power Sources. 2000;91(2):83-5.<br />

23. Zhang JG, Bruce PG, Zhang XG. Metal-air batteries. In:<br />

24. Daniel C, Besenhard JO, editors. Handbook of battery<br />

materials. Weinhiem (Germany): Wiley-VCH Verlag & Co.<br />

KGaA; 2011. p. 759-68.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):305-314.<br />

DOI: 10.7162/S1809-977720130003000011<br />

Contribution of audiovestibular tests to the topographic diagnosis of<br />

sudden deafness<br />

Jeanne Oiticica 1 , Roseli Saraiva Moreira Bittar 2 , Claudio Campi de Castro 3 , Signe Grasel 4 , Larissa Vilela Pereira 5 ,<br />

Sandra Lira Bastos 5 , Alice Carolina Mataruco Ramos 5 , Roberto Beck 5 .<br />

1) MD PhD. Department of Otolaryngology, University of São Paulo School of Medicine.<br />

2) MD PhD. Department of Otolaryngology, University of São Paulo School of Medicine.<br />

3) MD PhD. Heart Institute [InCor], University of São Paulo School of Medicine.<br />

4) MD PhD. Department of Otolaryngology, University of São Paulo School of Medicine.<br />

5) MD. Department of Otolaryngology, University of São Paulo School of Medicine.<br />

Institutions: (A) Department of Otolaryngology, University of São Paulo School of Medicine.<br />

(B) Heart Institute (InCor), University of São Paulo School of Medicine.<br />

São Paulo / SP - Brazil.<br />

Mailing address: Jeanne Oiticica, M.D., Ph.D. ENT Assistant Doctor Department of Otolaryngology University of São Paulo School of Medicine - R. Marjorie Prado<br />

160 - São Paulo / SP - Brazil - Zip Code: 04663-080 - E-mail: jeanneoiticica@bioear.com.br<br />

Article received on February 27 th , 2013. Article accepted on March <strong>17</strong> th , 2013.<br />

SUMMARY<br />

Introduction: Sudden hearing loss (SHL) is an ENT emergency defined as sensorineural hearing loss (SNHL) > 30 dB HL<br />

affecting at least 3 consecutive tonal frequencies, showing a sudden onset, and occurring within 3 days. In cases of SHL, a<br />

detailed investigation should be performed in order to determine the etiology and provide the best treatment. Otoacoustic<br />

emission (OAE) analysis, electronystagmography (ENG), bithermal caloric test (BCT), and vestibular evoked myogenic potential<br />

(VEMP) assessments may be used in addition to a number of auxiliary methods to determine the topographic diagnosis.<br />

Objective: To evaluate the contribution of OAE analysis, BCT, VEMP assessment, and magnetic resonance imaging (MRI) to<br />

the topographic diagnosis of SHL.<br />

Method: Cross-sectional and retrospective studies of 21 patients with SHL, as defined above, were performed. The patients<br />

underwent the following exams: audiometry, tympanometry, OAE analysis, BCT, VEMP assessment, and MRI. Sex, affected side,<br />

degree of hearing loss, and cochleovestibular test results were described and correlated with MRI findings. Student’s t-test was<br />

used for analysis of qualitative variables (p < 0.05).<br />

Results: The mean age of the 21 patients assessed was 52.5 ± 15.3 years; 13 (61.9%) were women and 8 (38.1%) were men.<br />

Most (55%) had severe hearing loss. MRI changes were found in 20% of the cases. When the audiovestibular test results were<br />

added to the MRI findings, the topographic SHL diagnosis rate increased from 20% to 45%.<br />

Conclusion: Only combined analysis via several examinations provides a precise topographic diagnosis. Isolated data do not<br />

provide sufficient evidence to establish the extent of involvement and, hence, a possible etiology.<br />

Keywords: Deafness; Hearing Loss, Sudden; Diagnosis; Vestibular Function Tests; Hearing Tests.<br />

INTRODUCTION<br />

Sudden hearing loss (SHL) is an ENT emergency<br />

that was first described in 1944 (1), but remains poorly<br />

understood. According to the National Institute for Deafness<br />

and Communication Disorders (NIDCD), it can be defined<br />

as any sensorineural hearing loss (SNHL) > 30 dB affecting<br />

at least 3 consecutive frequencies, showing a sudden<br />

onset, and occurring within 3 days (2).<br />

The incidence of SHL is 5–20 per 100,000 population<br />

per year in the United States (3), 8 per 100,000 population<br />

per year in Thailand (4), and 27.5 per 100,000 population<br />

per year in Japan (5), where a increase has been observed<br />

over the past 30 years. The natural course leads to<br />

spontaneous recovery in 45–65% of the cases (6-8).<br />

Among patients undergoing drug therapy, recovery rates<br />

vary between 50% and 78% (5, 9). About half of the<br />

patients undergoing treatment recover hearing within the<br />

first few days and the other half within 3 months, but a small<br />

percentage show delayed recovery (9).<br />

Despite its sudden onset, which is often associated<br />

with symptoms such as dizziness and tinnitus, the<br />

pathophysiology of this condition remains undefined.<br />

Routine tests for diagnostic evaluation fail to detect the<br />

etiology in up to 88% of the cases (10). The list of<br />

potentially causative or associated agents is long; many of<br />

these are etiologic factors such as vestibular nerve<br />

schwannoma, infections, stroke, and neoplastic lesions,<br />

whereas others are only associated factors for which a<br />

causal relationship remains to be established (viral,<br />

autoimmune, vascular mechanisms) (11-13). Early diagnosis<br />

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and therapeutic management improve the patients’ quality<br />

of life.<br />

Tinnitus and aural fullness are reported in 95% of the<br />

patients with SHL, whereas dizziness is present in 55% of<br />

the cases showing the involvement of the posterior labyrinth<br />

and suggesting a worse prognosis (14). A detailed<br />

investigation of patients with SHL should be performed in<br />

all cases, the main objective of which should be to<br />

determine the etiology of the event in an effort to provide<br />

the best treatment for that patient. Furthermore, clinical<br />

evaluation may provide additional new data and a more<br />

detailed overview of the prognosis of patients with<br />

idiopathic SHL.<br />

Recent studies have demonstrated the role of tests<br />

such as otoacoustic emission (OAE) analysis,<br />

electronystagmography (ENG), bithermal caloric test (BCT),<br />

and vestibular evoked myogenic potential (VEMP)<br />

assessment in indicating disease prognosis, and also<br />

elucidated various auxiliary methods for determining the<br />

topographic diagnosis (15-<strong>17</strong>).<br />

OAE analysis is an important test for objective<br />

assessment of the functioning of the inner ear, specifically<br />

the outer hair cells (OHCs) of the cochlea. OAEs are lowintensity<br />

acoustic signals generated by nonlinear mechanical<br />

activity of the OHCs of the <strong>org</strong>an of Corti (18). OAEs can<br />

occur spontaneously (spontaneous OAEs) or in response<br />

to acoustic stimuli (evoked OAEs), which, once amplified,<br />

can be detected in the external auditory canal (EAC) (19).<br />

The sound stimulus reaches the cochlea and induces<br />

vibration of the basilar membrane, which in turn causes<br />

deflection of OHC stereocilia, ion flow, a voltage difference,<br />

and subsequent contraction of these cells to generate<br />

electromotility, which is thought to be responsible for the<br />

phenomenon of OAEs. The presence of OAEs indicates<br />

that the conductive mechanisms of the ear (external ear<br />

canal, tympanic membrane, and ossicular chain) and the<br />

OHCs are functioning properly, and therefore, in any kind<br />

of hearing loss (HL), OAE assessment is a valuable step.<br />

However, we must remember that OAEs do not provide<br />

information about the inner hair cells (IHC), the eighth<br />

cranial nerve, or the ascending auditory pathways.<br />

Spontaneous OAEs reveal narrow band acoustic energy<br />

from the cochlea regardless of the presence of a sound<br />

stimulus. They are of little clinical importance because they<br />

are present in only 40–60% of subjects with normal hearing<br />

(20). Evoked OAEs can be classified as transient-evoked<br />

otoacoustic emissions (TEOAEs) and distortion-product<br />

otoacoustic emissions (DPOAEs). TEOAE responses are<br />

caused by acoustic stimuli, usually clicks, but frequencyspecific<br />

stimuli such as tone bursts and tone pips can also<br />

induce these responses. Although a click is a broadband<br />

stimulus that activates the whole cochlea, TEOAEs can<br />

provide the “frequency-specific” pattern of the cochlea.<br />

They can be divided into frequency bands representing<br />

responses from different segments of the cochlea (21).<br />

DPOAEs are generated in the cochlea in response to the<br />

simultaneous presentation of 2 pure tones (f1 and f2<br />

stimuli). The cochlea has nonlinear properties that produce<br />

changes in the output signal that are not directly related to<br />

the input signal, creating responses at frequencies other<br />

than those provided by the 2 input signals. These responses<br />

are called distortion products and indicate normal activity<br />

of the inner ear. The 2f1-f2 ratio is the most commonly<br />

used as it results in the most robust and reliable responses.<br />

DPOAEs are present in almost all subjects with normal<br />

hearing thresholds. OAEs have been widely used as an<br />

objective and non-invasive screening method for HL. OAEs<br />

can still be useful in the differential diagnosis of cochlear<br />

and retrocochlear HL. As they arise from OHCs (peripheral<br />

auditory system), responses are supposed to be compatible<br />

with auditory thresholds in the case of sensory HL. In the<br />

case of retrocochlear pathology, DPOAE responses may be<br />

present even with thresholds worse than 45 dB HL and<br />

abnormal ABR since DPOAEs reflect pre-neural sensory<br />

activity from the cochlea. However, this is an uncommon<br />

finding. Studies indicate that about 20% of patients with a<br />

retrocochlear pathology have normal DPOAEs. Expanding<br />

lesions in the internal auditory canal (IAC) or posterior fossa<br />

may decrease the cochlea blood flow and affect the<br />

presence of OAEs. In SHL, the OAE test is a fast and elegant<br />

method for confirming HL, excluding any psychogenic<br />

deafness, and monitoring treatment outcome (<strong>17</strong>).<br />

The ENG permits electrical recording of eye<br />

movements and is the most common method used for the<br />

diagnosis of peripheral vestibular disorders. The caloric test<br />

allows for independent assessment of the right and left<br />

labyrinths (horizontal semicircular canals) in response to<br />

bithermal irrigation of the external auditory canal. Each ear<br />

is irrigated twice to elicit both excitatory and inhibitory<br />

responses. Thus, the caloric test provides a functional<br />

assessment of the horizontal semicircular canal and superior<br />

vestibular nerve, contributing to the topographic study<br />

of the peripheral <strong>org</strong>an and ascending vestibular pathways.<br />

The VEMP assessment is a newly developed vestibular<br />

test that is used clinically to analyze otolith function<br />

and is the only specific test for the inferior vestibular nerve.<br />

In primitive vertebrates, the saccule is not part of the<br />

vestibular system but is a structure of the auditory system.<br />

In most vertebrates and humans, the saccule is sensitive to<br />

acoustic stimuli but is involved with vestibular function.<br />

Furthermore, as a noninvasive and low-risk test, the VEMP<br />

assessment does not depend on cochlear integrity and may<br />

be present in patients with profound deafness. This potential<br />

can be evoked by a sound stimulus and recorded in the<br />

cervical region. In this way, VEMP analysis assesses the<br />

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integrity of the sacculocollic reflex, which depends on the<br />

functional integrity of the macula of the saccule, inferior<br />

vestibular nerve, lateral vestibular nucleus, descending<br />

vestibulospinal tract, and the accessory nerve to the<br />

sternocleidomastoid muscle (SCM) and its neuromuscular<br />

junction. Therefore, unlike other exams, VEMP assessment<br />

enables the study of structures not commonly assessed by<br />

traditional vestibular tests (the saccule and inferior vestibular<br />

nerve) as well as the descending and ascending vestibular<br />

pathways. Lesions anywhere in this pathway can<br />

result in abnormal test results. The literature on VEMP in<br />

patients with SHL remains very controversial. Some authors<br />

describe a positive correlation between the presence of<br />

normal VEMP and a good prognosis for hearing recovery in<br />

patients with SHL (22, 23). Other authors have noted<br />

normal VEMP results in all patients with SHL (24), or did not<br />

relate these to hearing outcome. However, VEMP<br />

assessment can contribute to the diagnosis of different<br />

neurotologic diseases, including Meniere’s disease, superior<br />

semicircular canal dehiscence, benign paroxysmal<br />

positional vertigo (BPPV), vestibular neuritis, and vestibular<br />

schwannoma, and it is a great method for topographic<br />

diagnosis. It evaluates not only the neural structures, but<br />

also the sensory structures of the saccule, which are<br />

sensitive and responsive to acoustic stimuli, although they<br />

do not contribute to hearing. For example, in a case of SHL<br />

of vascular origin, the lesion is expected to be large and<br />

involve not only the cochlea, but also the vestibule,<br />

damaging a greater number of sensory-neural structures. In<br />

such cases, combined analysis of audiological and<br />

electrophysiological test results and imaging studies may<br />

contribute directly to the topographic diagnosis of the<br />

lesion and to the subsequent prognosis of the patient’s<br />

SHL. Currently, the role of these diagnostic methods in the<br />

field of SHL remains uncertain, and more studies are<br />

needed to better determine the practical implications of<br />

these tests.<br />

OBJECTIVE<br />

To evaluate the contribution of OAE analyses, the<br />

bithermal caloric test, VEMP assessments, and magnetic<br />

resonance imaging (MRI) to the topographic diagnosis of<br />

SHL.<br />

METHOD<br />

This cross-sectional study included patients with a<br />

history of SHL, as defined above, who consulted the<br />

Department of Neurotology, Hospital das Clínicas, University<br />

of São Paulo School of Medicine (HC-FMUSP) from January<br />

2011 to January 2012. This study was previously approved<br />

by the Hospital’s Ethics Committee on Research (1<strong>17</strong>9/<br />

07). All patients followed the outpatient care protocol for<br />

SHL, including providing a detailed history, and undergoing<br />

an ENT physical examination as well as the following<br />

exams: pure tone and speech audiometry, tympanometry,<br />

DPOAE analysis, ENG, VEMP assessment, laboratory tests,<br />

and MRI. All tests were performed at the University of São<br />

Paulo School of Medicine.<br />

Inclusion criteria<br />

We included all patients who satisfied the following<br />

criteria:<br />

• Were diagnosed with SHL (sensorineural hearing loss ><br />

30 dB over at least 3 contiguous frequencies developed<br />

within 72 hours) and confirmed by pure tone audiometry;<br />

for patients who had no prior audiometry results, the<br />

audiometry findings of the contralateral side were<br />

considered representative of the original auditory<br />

threshold.<br />

• Performed the DPOAE test.<br />

Exclusion criteria<br />

We excluded patients who met any of the following<br />

criteria:<br />

• Did not agree to participate in the study<br />

• Failed to comply with the follow-up procedure or who<br />

had contraindications for any test (metallic objects<br />

implanted in the body preventing the realization of<br />

MRI, tympanic membrane perforation preventing the<br />

completion of ENG with water, middle ear effusion,<br />

otosclerosis or ossicular chain disjunction preventing<br />

DPOAE or VEMP assessments, as these conditions<br />

would interfere with the outcome).<br />

Pure tone and speech audiometry,<br />

tympanometry<br />

Air- and bone-conducted pure tone thresholds were<br />

obtained in the frequency range of 250 to 8000 Hz and 500<br />

to 4000 Hz, respectively, in addition to speech recognition<br />

threshold (SRT) and word recognition scores (WRS). When<br />

the patient showed no speech discrimination at the highest<br />

intensity permitted by the equipment, we tested the<br />

speech detection threshold (SDT). Immitance<br />

measurements included tympanometry and contralateral<br />

acoustic reflex thresholds at 500 to 4000Hz. The reflex was<br />

considered present when detected for at least 1 of 4<br />

frequencies and absent when no response was detected<br />

for any frequency. The PTA (Pure Tone Average) was<br />

obtained by averaging the pure tone thresholds of 0.5, 1,<br />

2, 4, 6, and 8 kHz.<br />

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Otoacoustic emissions - OAE<br />

Before the OAE test, an otoscopy was performed to<br />

prevent cerumen or debris from blocking the probe (21).<br />

To ensure proper acquisition of OAEs, the test was<br />

performed in a quiet environment with a noise level < 40dB<br />

HL. The patient was instructed to relax and avoid jaw<br />

movements, swallowing, or voice emissions that could<br />

generate noise in the external auditory canal (21). DPOAEs<br />

were tested at the intensities of 65 dB HL (f1) and 55 dB<br />

HL (f2) in the frequency range 750 to 8000 Hz. A response<br />

was considered to be significant when the signal-to-noise<br />

ratio was > 6dB. When significant responses were obtained<br />

for 5 or more frequencies, DPOAEs were considered to be<br />

present. In this case, further analysis was undertaken to<br />

determine whether DPOAEs were consistent with auditory<br />

thresholds. Present responses showed proper cochlear<br />

function of OHCs at these frequencies. We used SCOUT<br />

software (Natus Medical Incorporated, Mundelein, Il, USA)<br />

for these analyses.<br />

Bithermal Caloric Test - BCT<br />

Before the BCT analysis, the patient underwent a<br />

standard ENG test battery with the aim of excluding other<br />

functional findings that could influence the post-caloric<br />

response. The test sequence consisted of recordings of eye<br />

movements in response to stimuli: (1) saccade test, (2)<br />

spontaneous nystagmus with open eyes (SNEO), (3)<br />

smooth pursuit (SP), (4) optokinetic test (OPK), (5) static<br />

position tests – supine, right ear down, left ear down, Rose,<br />

sitting, body right and body left, (6) BCT. For the BCT, the<br />

patient was in a supine position with the head bent forward<br />

30 o and eye movements were recorded after irrigation with<br />

water for 40 seconds in the following sequence: (a) warm<br />

irrigation at 44 o C, left ear, (b) warm irrigation at 44 o C, right<br />

ear, (c) cool irrigation at 30 o C, left ear, (d) cool irrigation at<br />

30 o C, right ear. In the BCT analysis, we considered nystagmus<br />

of both labyrinths, measured through the angular velocity<br />

of the slow-phase velocities (SPV). We observed the<br />

nystagmus direction, rhythm, amplitude, and frequency,<br />

and compared the functioning of the labyrinths. The SPV<br />

measure was provided by the computer system and was<br />

considered normal between 7 and 50 o /second. The results<br />

were classified according to the post-caloric values: (1)<br />

normal: post-caloric responses within these limits, (2)<br />

hyperactivity: post-caloric responses exceeding 50 o /second,<br />

(3) hypoactivity: post-caloric responses below 7 o /second,<br />

and (4) no response: complete absence of caloric responses<br />

after stimulation. To evaluate the response symmetry<br />

between the vestibules, 2 values were calculated: (a)<br />

Unilateral Weakness (UW) and (b) Directional<br />

Preponderance (DP). UW shows the relative difference<br />

between the right and left ear responses, and DP shows the<br />

relative difference of right beating vs. left beating nystagmus<br />

directions. We considered UW up to 18% and DP up to 20%<br />

as reference values (25). UW has a high clinical value and<br />

indicates a vestibular asymmetry of peripheral or central<br />

origin; it always points to the weaker ear. DP indicates the<br />

stronger nystagmus direction. We considered signs of<br />

central origin to include the absence of the inhibitory effect<br />

of ocular fixation suppression (FS) and reversal of the<br />

direction of post-caloric nystagmus.<br />

Vestibular evoked myogenic potentials -<br />

VEMP<br />

A cervical VEMP assessment was performed using<br />

AEP software, version 7.0.0, Bio-logic Navigator Pro system<br />

(Natus Medical Incorporated, Mundelein, Il, USA). During<br />

the test, the patient remained comfortably seated with neck<br />

rotation contralateral to the sound stimulation. The sound<br />

stimuli were presented through insert earphones (ER-3)<br />

calibrated according to ANSI S1.40-1984 (American National<br />

Standard Institute, 2001). The surface electrodes were<br />

placed on the forehead (ground), the upper part of the<br />

sternum (negative), and the upper third of the contracted<br />

SCM muscle (positive electrode). The impedance was 40% (with the<br />

lowest amplitude from the SHL side) or the absence of a<br />

response on the affected side, suggesting saccule and<br />

inferior vestibular nerve involvement.<br />

Magnetic resonance imaging (MRI) of<br />

the inner ear and brain stem<br />

MRI of the inner ear was carried out by spin-echo<br />

and fast spin-echo sequences in T1 and T2, with multiplanar<br />

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acquisition before and after intravenous administration of<br />

the paramagnetic contrast agent (gadolinium). FIESTA<br />

series were also included in volume acquisition with cuts of<br />

0.8 mm. MRI was considered abnormal only when findings<br />

were obviously related to SHL, such as schwannoma of the<br />

eighth cranial nerve (intracanalicular or cerebellopontine<br />

angle), internal carotid artery obstruction, stroke, and<br />

cochleovestibular lesions (inflammation or vascular), as<br />

previously described (26). All other MRI findings were<br />

described as normal, including those whose etiological<br />

association with SHL were possible but not previously<br />

proven (vascular anomalies, demyelinating diseases) or<br />

unknown (white matter changes, vascular loops,<br />

contralateral cochlear abnormalities, enhancement of<br />

mastoid cells or the endolymphatic duct, ventricular<br />

dilatation, asymmetry of the cerebellum).<br />

Study variables<br />

Study variables included the following: (a) categorical<br />

variables (CVs), and (b) quantitative variables (QVs). The<br />

CVs were described by their frequency distribution, and<br />

included the following variables: (1) sex; (2) the affected<br />

side; (3) classification of the degree of HL according to pure<br />

tone thresholds (up to 40 dB HL mild, 40–70 dB HL<br />

moderate, 70–90 dB HL severe, and >90 dB HL profound)<br />

(27). We considered anacusis as the complete absence of<br />

responses at all frequencies; (4) The presence or absence<br />

of the acoustic reflex during immitance measurements; (5)<br />

the presence or absence of DPOAE responses; (6) the<br />

presence of normal reflexes, hyperfunction, hypofunction,<br />

or no responses in the BCT; (7) normal or abnormal VEMP<br />

responses; and (8) normal and abnormal MRI findings. The<br />

QV were described by mean and standard deviation, and<br />

included (1) age; (2) PTA; (3) WRS.<br />

Statistical analysis<br />

CV results were described based on their frequency.<br />

QV results were expressed as the mean and standard<br />

deviation. Comparisons of QV were calculated using an<br />

unpaired t-test. The level of statistical significance was set<br />

at 5% (p < 0.05). We also calculated the 95% confidence<br />

interval of the mean and verified whether the 2 QV had<br />

equal standard deviations. Statistical analyses were<br />

performed using the GraphPadInstat program.<br />

RESULTS<br />

Between January 2011 and January 2012, 40 patients<br />

diagnosed with SHL were treated at the Department of<br />

Neurotology, Hospital das Clínicas, University of São Paulo<br />

15%<br />

Figure 1. Classification of NSHL in patients with SHL.<br />

School of Medicine. Twenty-one of these had DPOAEs and<br />

constituted our final sample. No patient refused to participate<br />

in the study, had contraindications to any of the exams, or<br />

missed their follow-up visits. The analysis of the 21 patients<br />

who met the selection criteria showed that the mean age<br />

and standard deviation was 52.5 ± 15.3 years, and that 13<br />

(61.9%) were females and 8 (38.1%) males. The right side<br />

was affected in 14 (66.6%) patients and the left side in 7<br />

(33.4%).<br />

Pure tone and speech audiometry,<br />

tympanometry<br />

Of the 21 enrolled patients, 20 underwent audiometry<br />

at the beginning and end of the study, only 1 patient only<br />

underwent the initial audiometry tests. Figure 1 shows the<br />

distribution of the degree of NSHL observed in 20 patients<br />

at the beginning of the analysis. The initial mean PTA and<br />

standard deviation was 80.5 ± 25.7 dB HL (n = 20), with a<br />

95% confidence interval of the mean between 68.5 and<br />

92.6 dB. The mean PTA at the end of the study and the<br />

standard deviation was 57.6 ± 34.3 dB HL (n = 20), with a<br />

95% confidence interval of the mean between 41.5 and<br />

73.7dB. The difference between the initial and final PTA<br />

was statistically significant (p = 0.0221, unpaired t-test),<br />

indicating that, in general, there was an improvement in<br />

the thresholds during follow-up due to treatment of SHL.<br />

ENG and PC<br />

15% 15%<br />

55%<br />

Moderate<br />

Severe<br />

Profound<br />

Anacusis<br />

Nineteen of the 21 patients (90.5%) had ENG with<br />

caloric tests. Two (9.5%) failed to show up for the test. Of<br />

the 19 patients on whom the tests were performed, 8<br />

(42.1%) had normal and 11 (57.9%) abnormal findings.<br />

One subject had DP on the same side as the SHL. The<br />

distribution of the remaining 18 test results of the affected<br />

ears can be seen in Figure 2.<br />

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Oiticica et al.<br />

22%<br />

36%<br />

45%<br />

Normal<br />

11%<br />

Hypoactivity<br />

Normal<br />

64%<br />

22%<br />

Hyperactivity<br />

Abnormal<br />

No response<br />

Figure 2. Distribution of bithermal caloric test responses in<br />

patients with SHL.<br />

Figure 3. Distribution of VEMP responses in patients with<br />

SHL.<br />

14%<br />

86%<br />

Present DPOAE<br />

Absent DPOAE<br />

Figure 5. Expansive lesion inside the right IAC.<br />

Figure 4. Distribution of DPOAE results in patients with SHL.<br />

VEMP<br />

Fourteen of 21 patients (66.6%) completed the<br />

VEMP test. Nine of these (64.3%) had normal tests. Of the<br />

5 (35.7%) with abnormal test results, 3 had an IADR > 40%,<br />

with the lowest amplitude response on the SHL side in all<br />

cases; 2 had no responses on the affected side (Figure 3).<br />

DPOAE<br />

All 21 patients included in the study completed<br />

DPOAE assessments. Only 3 (14.3%) showed present<br />

DPOAEs at most frequencies. Although DPOAE were<br />

requested at the time of initial evaluation, in view of the<br />

test schedules and the demand of the institution, some of<br />

these tests were eventually completed during follow-up of<br />

SHL after partial or complete recovery of NSHL, and the<br />

results were consistent with the audiometric thresholds<br />

obtained at the same time as the DPOAE tests. In the<br />

remaining 18 (85.7%) patients, DPOAEs were considered<br />

absent according to our criteria (Figure 4).<br />

MRI<br />

MRI was performed in 20 (95.2%) of 21 patients as<br />

1 patient failed to complete the test. The MRI findings<br />

were normal in 16 (80%) patients and abnormal in 4 (20%).<br />

Analyzing the 4 abnormal tests, we found that in 2 cases the<br />

lesion arose from the eighth cranial nerve (schwannoma)<br />

ipsilateral to the SHL. In case 9, MRI revealed an expansive<br />

lesion inside the right IAC, protruding into the ipsilateral<br />

cerebellopontine angle (CPA) (Figure 5). In case 11, MRI<br />

showed an expansive lesion located in the right CPA and<br />

IAC, with enlargement and partial obliteration of the<br />

ipsilateral cistern (Figure 6). Among the 2 patients with<br />

eighth cranial nerve schwannomas, DPOAEs were absent<br />

in 1 (case 9) but present in the other (case 11). In this case,<br />

the test was performed later, during follow-up of SHL, and<br />

after complete recovery of audiometric hearing thresholds.<br />

Similarly, VEMP was absent in case 9 and present in case 11.<br />

ENG showed hypofunction in case 9 and no caloric<br />

function at all in case 11. In the other 2 cases with abnormal<br />

MRIs, the lesion was located in the cochlea on the same<br />

side as the SHL. The lesion was described by the radiologist<br />

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as a focus of post-contrast enhancement in the cochlea,<br />

vestibule, and ipsilateral semicircular canals suggestive of<br />

inflammation (viral labyrinthitis, fibrotic tissue) in 1 (case<br />

6), and as enhancement of the ipsilateral cochlea (possible<br />

inflammation) in the other (case 18). In the 2 latter cases,<br />

DPOAEs were absent, which was in agreement with the<br />

MRI findings showing cochlear enhancement, and helped<br />

to prove that the auditory lesion was peripheral, involving<br />

the OHCs, and not neural. VEMP was abnormal in case 6<br />

and was not assessed in case 18. Thus, ENG showed no<br />

vestibular function ipsilateral to the lesion in both cases,<br />

even without any sign on MRI of a lesion extending into the<br />

posterior labyrinth in case 18.<br />

We also evaluated the mean difference between<br />

the initial and final PTA to compare the average hearing<br />

recovery among patients whose topographic diagnosis<br />

showed smaller, more restricted lesions (absent or Cn or<br />

SVn lesions) or more extensive lesions (Cn + VSn or Cn +<br />

VSn + IVn). The mean PTA recovery and standard deviation<br />

Figure 6. Expansive lesion in the right IAC and CPA with<br />

widening and partial obliteration of the ipsilateral CPA cistern.<br />

Table 1. Test results for all 21 subjects.<br />

Case Side Audiometry I DPOAE BCT VEMP MRI Topographic Diagnosis<br />

Initial PTA Final PTA<br />

(affected nerve)<br />

(dB HL) (dB HL)<br />

1 R 75.8 73.3 NP - h NP nl Cn<br />

2 L 80.8 25 + - nl nl nl Cn<br />

3 R 50.8 24 - + ¯ nl nl SVn<br />

4 L 59 NP + - NP NP nl Cn<br />

5 R 86.7 79 NP - ¯ Abn nl Cn + SVn + IVn<br />

6 R 47.5 29 NP - 0 Abn Abn* Cn + SVn + IVn<br />

7 R 81.6 25 NP + nl nl nl -<br />

8 L 120 95,8 NP - 0 Abn nl Cn + SVn + IVn<br />

9 R 120 120 NP - ¯ Abn Abn** Cn + SVn + IVn<br />

10 R 64 35 NP - ¯ nl NP Cn + SVn<br />

11 R 37.5 10 + 0 nl Abn*** SVn<br />

+<br />

12 R 70,8 64,1 NP - nl nl nl Cn<br />

13 R 69 25,8 + - nl NP nl Cn<br />

14 R 65 46 NP - h nl nl Cn + SVn<br />

15 L 67.5 81 - - nl NP nl Cn<br />

16 R 81.6 22,5 - - nl NP nl Cn<br />

<strong>17</strong> L 1<strong>17</strong>.5 120 NP - DP Abn nl Cn + SVn + IVn<br />

18 L 120 120 NP - 0 NR Abn**** Cn + SVn<br />

19 L 83.3 47.5 NP - nl nl nl Cn<br />

20 R 120 76.6 NP - NR NR nl Cn<br />

21 R 112.5 104 NP - nl nl nl Cn<br />

I: immitance measurements; DPOAE: distortion-product otoacoustic emissions; BCT: bithermal caloric test; VEMP: vestibular evoked<br />

myogenic potential; MRI: magnetic resonance imaging; R: Right, L: left; dB: decibels; -: absent, +: present; : hyperactivity; : hypoactivity;<br />

nl: normal; 0: no response, DP: directional preponderance; NP: not performed; Cn: cochlear nerve; SVn: superior vestibular nerve; IVn:<br />

inferior vestibular nerve; Abn: Abnormal, -: none; *: Spotlights of post-contrast enhancement in the cochlea, vestibule, and right HSC that<br />

could represent inflammation (viral labyrinthitis, fibrotic tissue); **: expansive lesion within the right internal auditory canal protruding into<br />

the ipsilateral cerebellopontine angle; ***: expansive lesion in the right internal auditory canal and cerebellopontine angle with widening and<br />

partial obliteration of the ipsilateral cistern; ****: area of enhancement in the left cochlea (inflammation?).<br />

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was 29.3 ± 23.8 dB (n = 12), with a 95% confidence interval<br />

of the mean between 14.1 and 44.5 dB in patients with<br />

topographically more restricted lesions. In patients with<br />

topographically more extensive lesions the mean PTA<br />

recovery and standard deviation was 13.2 ± 10.9 dB (n =<br />

8) with a 95% confidence interval of the mean between 4<br />

and 22.4 dB. Case 4 was excluded from the analysis<br />

because only the initial audiometry results were available<br />

and no differences between the initial and final PTA could<br />

be calculated. The difference in the mean PTA recovery<br />

among patients with topographically more restricted lesions<br />

was statistically significantly different (p = 0.05, unpaired<br />

t-test) from those with more extensive lesions, indicating<br />

that patients with more extensive lesions show poorer<br />

hearing recovery.<br />

DISCUSSION<br />

In this cross-sectional study of 21 patients with SHL,<br />

abnormal MRI findings were seen in 20% of cases, which is<br />

similar to the results of a previous report on SHL at our<br />

institution that showed abnormal MRI findings in 25% of<br />

those studied (28). The small difference in abnormal MRI<br />

findings may be due to the smaller sample size used in this<br />

study. We chose to use DPOAE as an inclusion criterion<br />

because the test permits the evaluation of each frequency<br />

separately and responses may be present with hearing<br />

thresholds up to 45 dB HL, whereas TEOAEs are usually<br />

absent when the thresholds exceed 20–30 dB HL (29).<br />

Therefore, this test is not only important for the differential<br />

diagnosis of peripheral and central lesions, but also because<br />

absent responses in the initial SHL evaluation exclude the<br />

possibility of psychogenic deafness (PD) or simulation.<br />

Psychogenic deafness (PD) is a type of conversion<br />

disorder and can be defined as HL that cannot be explained<br />

by <strong>org</strong>anic lesions or anatomical or physiological changes.<br />

In such cases, the patient’s clinical history reveals<br />

psychological factors that could act as a trigger at the<br />

beginning of the event. It is possible to establish the<br />

diagnosis by comparing behavioral (pure tone audiometry)<br />

and objective tests (ABR and DPOAE), as these usually<br />

show divergent results: patients typically exhibit elevated<br />

pure tone thresholds but normal ABR and DPOAE responses.<br />

Attention is drawn to case 7 in Table 1. This patient<br />

presented with initial severe HL although tests failed to<br />

show any evidence of a structural lesion and follow-up<br />

showed a full recovery. One hypothesis for this case is PD.<br />

Malingering may also lead to divergent results between<br />

behavioral and objective tests, but in contrast to PD, it is<br />

usually is associated with an inconsistent history, personal<br />

advantages associated with deafness, and differences<br />

between pure tone audiometry and SRT >15 dB, which can<br />

be detected by an experienced audiologist. Therefore, in<br />

routine clinical evaluation of patients with SHL, DPOAE is<br />

a valid tool and contributes to diagnostic accuracy. If an<br />

unexpected response is present during the initial evaluation,<br />

the possibility of a retrocochlear lesion should be considered<br />

and investigated.<br />

In our study, DPOAEs were eventually performed<br />

late during follow-up of SHL and not as part of the initial test<br />

as planned. When performed during follow-up, it may be<br />

useful for showing possible cochlear recovery. This can<br />

happen even if the cause of SHL is an expansive lesion of<br />

the IAC or PCA, provided cochlear blood flow is preserved<br />

and cochlear integrity is ensured. In case 11, normal VEMP<br />

and DPOAE test results suggested normal saccular, inferior<br />

vestibular nerve, and cochlear (OHC) function. Pure tone<br />

and speech audiometry results were normal at the time the<br />

DPOAE was performed, indicating complete hearing<br />

recovery after initial moderate HL. We speculate that the<br />

initial hearing impairment caused by the expansive lesion<br />

extending from the IAC to CPA may have improved as a<br />

result of decreased edema resulting in a favorable treatment<br />

outcome. In the same case, the absence of a right vestibular<br />

response, as shown by BCT, suggests that only the superior<br />

vestibular nerve was affected.<br />

In case 9, the lesion was primarily located in the<br />

right IAC (although protruding into the CPA), and the test<br />

results (complete deafness on PTA, absent DPOAEs,<br />

abnormal VEMP, hypoactivity in BCT) confirmed that the<br />

whole cochleo-vestibular system was affected on this side.<br />

Therefore, in our view, DPOAE analysis is an important test<br />

for SHL evaluation since it is the only one that assesses<br />

cochlear function.<br />

Patients underwent a complete ENG evaluation in<br />

order to rule out other changes to vestibular-ocular function<br />

suggestive of central involvement. No abnormalities<br />

suggesting central dysfunction were found in our series.<br />

Therefore, we considered only the BCT results in our<br />

analysis. The importance of vestibular evaluation in SHL<br />

becomes clear when we look at the cases with abnormal<br />

MRIs. Cases 6, 9, 11, and 18 were found to have structural<br />

lesions by MRI and all had impaired superior vestibular<br />

nerve function, with absent vestibular responses in 3 (75%)<br />

of the 4 cases. Only 1 case had residual function, but<br />

hypoactivity (SPV below 7 º/sec), highlighting the<br />

importance of BCT for SHL prognosis. Overall, 4 cases in<br />

our sample showed no response, 3 (75%) had abnormal<br />

MRIs and 2 of them (50%) tumors, and so absent caloric test<br />

responses require an imaging study. However, abnormal<br />

imaging findings do not predict permanent deafness, since<br />

cases 6 and 11 both experienced hearing recovery.<br />

Audiovestibular function tests and MRI are important tools<br />

that together provide us with a complete overview of the<br />

functional impairment and the sites involved in SHL, and<br />

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they may also be helpful for establishing the hearing<br />

prognosis for these patients.<br />

MRI demonstrated structural changes in 20% of the<br />

patients (4 cases: 3 with an extensive lesion and 1 with a<br />

restricted lesion). When the audiovestibular test results<br />

were combined with the MRI findings, the proportion with<br />

a topographic SHL diagnosis increased from 20% to 45%.<br />

CONCLUSION<br />

Only combined analysis of several test results allows<br />

for a precise topographic diagnosis. Isolated test results do<br />

not provide sufficient data to establish the extent of SHL<br />

involvement and hence a possible etiology. The<br />

combination of all tests was more efficient for topographic<br />

diagnosis than just audiometry and MRI. Combined<br />

assessment permits a functional evaluation that cannot be<br />

obtained with MRI alone. Each test evaluates a different<br />

segment of the cochleo-vestibular system and permits an<br />

overview of the degree of functional or structural<br />

impairment. The auditory evoked potentials (ABRs) could<br />

be included in the audiovestibular test battery. ABR may be<br />

useful to rule out malingering and patients with psychogenic<br />

hearing loss, and may also be helpful in follow-up of SHL,<br />

especially in patients with schwannomas.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):315-320.<br />

DOI: 10.7162/S1809-977720130003000012<br />

Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy<br />

for botulinum toxin injection: an experimental study in cadaver larynges<br />

Valéria Maria de Oliveira Alonso 1 , Azis Arruda Chagury 2 , Adriana Hachiya 3 , Rui Imamura 3 , Domingos Hiroshi Tsuji 4 ,<br />

Luiz Ubirajara Sennes 4 .<br />

1) Otolaryngologist, PhD. Specialist in Otolaryngology, School of Medicine, University of São Paulo.<br />

2) Otolaryngologist. Specialist in Otolaryngology, School of Medicine, University of São Paulo.<br />

3) Otolaryngologist, PhD. Associate Doctor, Department of Otolaryngology, School of Medicine, University of São Paulo.<br />

4) Otolaryngologist, PhD. Professor in the Department of Otolaryngology, School of Medicine, University of São Paulo.<br />

Institution: Department of Otolaryngology, University of São Paulo School of Medicine.<br />

Av. Dr. Enéas de Carvalho Aguiar, 255, São Paulo / SP - Brazil - Zip Code: 05403-900<br />

Mailing address: Azis Arruda Chagury - Av. Dr. Enéas de Carvalho Aguiar, 255 - Instituto Central - São Paulo / SP - Brazil - Zip Code: 05403-900 - Telephone: (+55 11)<br />

2661-6286 - E-mail: azischagury@gmail.com<br />

Article received on March 4 th , 2013. Article accepted on April 8 th , 2013.<br />

SUMMARY<br />

Introduction: Endolaryngeal injection of botulinum toxin into the thyroarytenoid (TA) muscle is one of the methods for<br />

treatment of focal laryngeal dystonia. However, after treatment, there is variation in laryngeal configuration as well as the side<br />

effects reported by patients. As a consequence of the functional variability of results, it was hypothesized that botulinum toxin<br />

diffuses beyond the limits of the muscle into which it is injected.<br />

Objectives: After injection of botulinum toxin into the TA muscle for the treatment of focal laryngeal dystonia, patients differ<br />

in terms of laryngeal configuration and side effects. We hypothesized that this toxin diffuses from the target muscle to adjacent<br />

muscles.<br />

Method: The TA muscles of 18 cadaver larynges were injected with aniline blue (0.2 mL). After fixation in formaldehyde and<br />

nitric acid decalcification, the larynges were sectioned in the coronal plane and the intrinsic muscles were analyzed.<br />

Results: We found diffusion of aniline blue to the lateral cricoarytenoid muscle, cricothyroid muscle, and posterior cricoarytenoid<br />

muscle in 94.3%, 42.9%, and 8.6% of the cases, respectively. In terms of the degree of diffusion to adjacent muscles, we found<br />

no differences related to the size (height and width) of the TA muscle or to gender.<br />

Conclusions: Our findings suggest that diffusion of botulinum toxin from its injection site in the TA muscle to the lateral<br />

cricoarytenoid muscle is likely in most cases. On the other hand, diffusion to the cricothyroid muscle occurs in approximately<br />

half of cases and diffusion to the posterior cricoarytenoid muscle occurs in very few cases.<br />

Keywords: Larynx; Dysphonia; Botulinum Toxins; Diffusion; Biological Transport.<br />

INTRODUCTION<br />

Adductor spasmodic dysphonia is a disorder<br />

characterized by involuntary contractions of the adductor<br />

muscles of the larynx during phonation, resulting in strained,<br />

strangled, or forced voice quality, as well as intermittent<br />

voice breaks (1,2). The use of botulinum toxin for the<br />

treatment of spasmodic dysphonia was described by Blitzer<br />

in 1986 (3). Since this report, botulinum toxin injection has<br />

been the treatment of choice for spasmodic dysphonia in<br />

various centers worldwide. Botulinum toxin injection is a<br />

minimally invasive method that provides excellent functional<br />

results. The toxin blocks presynaptic acetylcholine release<br />

at the neuromuscular junction, causing a partial and reversible<br />

chemodenervation leading to muscle weakness, which<br />

reduces spasms in patients with dystonia (4).<br />

The most widely used method for injecting botulinum<br />

toxin into the thyroarytenoid (TA) muscle is<br />

electromyography-guided injection (4). At our facility,<br />

botulinum toxin is typically injected with a flexible needle,<br />

which is passed through the biopsy channel of a fiberoptic<br />

laryngoscope and guided by video-assisted endoscopy<br />

(2,5).<br />

The reported side effects of botulinum toxin injection<br />

into the TA muscle include dysphagia, aspiration, and<br />

transient dyspnea, effects that vary in terms of incidence<br />

and severity (6,10). Another factor observed in clinical<br />

practice is a change in glottal configuration, including vocal<br />

fold bowing and incomplete glottal closure (with or without<br />

ventricular fold paralysis).<br />

Studies in ophthalmology and gastroenterology have<br />

shown that paralysis of adjacent muscles is the most serious<br />

complication of the use of botulinum toxin and is probably<br />

caused by excessive diffusion of the toxin (11-13). This<br />

probably accounts for the wide variability in laryngeal<br />

function and configuration after injection of botulinum<br />

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toxin into the TA muscle, given that the adjacent (nontargeted)<br />

muscles include the lateral cricoarytenoid (LCA)<br />

muscle and the ventricular portion of the TA muscle itself.<br />

Therefore, the objective of the present study was to<br />

evaluate the diffusion of aniline blue, a stain specific to<br />

muscle fibers, after its injection into the TA muscles of<br />

larynges excised from human cadavers.<br />

METHOD<br />

This was an experimental study of 18 larynges that<br />

were excised from human cadavers. The larynges were<br />

obtained from the University of São Paulo School of<br />

Medicine, São Paulo Municipal Department of Death<br />

Certification, located in the city of São Paulo, Brazil. All<br />

larynges were removed within the first 24 h after death.<br />

We used 14 male larynges and 4 female larynges. All<br />

larynges were obtained from adult individuals of 30–87<br />

years of age (mean age, 55.5 years). The cause of death<br />

was not taken into consideration, given that all larynges<br />

were macroscopically intact.<br />

In the present study, we used aniline blue, a watersoluble<br />

stain that targets muscle fibers, at a concentration<br />

of 2%. The methodology employed was similar to that<br />

used for the injection of botulinum toxin in the Clinical<br />

Otolaryngology Department of the University of São<br />

Paulo School of Medicine Hospital das Clínicas (5). We<br />

used a flexible needle (model no. IN-2010; Machida<br />

Endoscope Co., Ltd., Tokyo, Japan) with a 0.5 mm tip.<br />

The needle was introduced into the TA muscle and<br />

middle third of the vibratory portion of the vocal fold,<br />

approximately 1 mm lateral to junction of the vibratory<br />

portion and the floor of the laryngeal vestibule (Figure 1).<br />

We injected 0.2 mL of aniline blue into the right and left<br />

vocal folds of each larynx. This volume (0.2 mL) is the<br />

volume that is used for botulinum toxin injections at our<br />

facility. The larynges remained in an anatomical position<br />

for 30 min, after which they were immersed in 10%<br />

buffered formalin and left for 7 days. The larynges were<br />

then placed in 7% nitric acid for 24 h for decalcification,<br />

being subsequently immersed in the same buffered<br />

formalin solution. The larynges were removed from the<br />

buffered formalin solution and stored at -10 o C. In order to<br />

standardize the laryngeal sections, we drew a line<br />

(corresponding to the projection of the right vocal fold)<br />

on the ipsilateral thyroid cartilage lamina, parallel to the<br />

lower border of the cartilage. After measuring the width<br />

of the lamina along this line, we divided the line into 4<br />

equal parts. The larynges were coronally sectioned with<br />

a microtome blade at 3 points (at the demarcated levels),<br />

resulting in 4 sections (designated sections I, II, III, and IV,<br />

from posterior to anterior). All sections were examined<br />

under a surgical microscope in order to identify the<br />

regions and muscles that showed aniline blue staining. For<br />

subsequent analyses, the sections were photographed<br />

with a digital camera (DSC-S30; Sony Corporation, Tokyo,<br />

Japan) at a resolution of 640 X 480 pixels.<br />

We examined the TA, LCA, cricothyroid (CT), and<br />

posterior cricoarytenoid (PCA) muscles for aniline blue<br />

staining. We examined both sides of the sections because<br />

of the possibility of finding different degrees of aniline<br />

blue staining at different depths. Stained muscles were<br />

defined as those in which the entire length of the muscle<br />

(in at least one of the sections) showed aniline blue<br />

staining. Partially stained muscles were defined as those<br />

in which part of the muscle, however small, did not show<br />

staining. In order to analyze the PCA muscle, we dissected<br />

the posterior lamina of the cricoid cartilage.<br />

We compared the genders in terms of dye diffusion<br />

to the muscles studied, each cadaver being considered as<br />

one case. Dye diffusion was defined as the presence of<br />

aniline blue staining on at least one side (right or left). We<br />

then measured the TA muscle in the section in which it<br />

was at its largest, using a ruler divided into millimeters.<br />

The TA muscle was measured in the lateromedial direction<br />

(width) and in the craniocaudal direction (height), as<br />

shown in Figure 2.<br />

In order to compare the muscles in terms of dye<br />

diffusion, we used the McNemar’s test for paired<br />

proportions. In order to compare the genders in terms of<br />

dye diffusion, we used Fisher’s exact test. In order to<br />

determine whether TA muscle width and height had any<br />

influence on dye diffusion to non-injected muscles, we<br />

adjusted the logistic regression model, testing the<br />

parameters with the Wald test. In order to compare the<br />

genders in terms of laryngeal size, we used the<br />

nonparametric Mann-Whitney test. For all tests, the level<br />

of significance was set at 5%.<br />

RESULTS<br />

We evaluated 18 larynges (with 36 vocal folds). The<br />

left vocal fold of one of the larynges was damaged during<br />

the sectioning process, and the larynx was therefore<br />

excluded from the study sample. Thus, our final sample<br />

consisted of 35 hemi-larynges. Table 1 shows the analysis<br />

of aniline blue staining of the TA, LCA, CT, and PCA<br />

muscles.<br />

Aniline blue staining was observed most often in the<br />

LCA muscle, followed by the CT and PCA muscles. We<br />

found statistically significant differences between the LCA<br />

and CT muscles and between the LCA and PCA muscles in<br />

terms of the frequency of dye diffusion (p < 0.00001). We<br />

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Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy for botulinum toxin injection: an experimental study in cadaver larynges.<br />

Alonso et al.<br />

Figure 2. Points at which the width and height of the<br />

thyroarytenoid muscle were measured.<br />

Figure 1. Introduction of the needle into the injection site.<br />

Table 1. Analysis of aniline blue staining of the thyroarytenoid, lateral cricoarytenoid,<br />

cricothyroid, and posterior cricoarytenoid muscles.<br />

Aniline Blue Staining<br />

Muscle No staining Partial staining Complete staining Any staining (total)<br />

n (%) n (%) n (%) n (%)<br />

TA - 2 (5.7%) 33 (94.3%) 35 (100%)<br />

LCA 2 (5.7%) 16 (45.7%) <strong>17</strong> (48.6%) 33 (94.29%)<br />

CT 20 (57.1%) 14 (40.0%) 1 (2.9%) 15 (42.86%)<br />

PCA 32 (91.4%) 3 (8.6%) - 3 (8.57%)<br />

Note: TA, thyroarytenoid; LCA, lateral cricoarytenoid; CT, cricothyroid; PCA,<br />

posterior cricoarytenoid.<br />

also found a statistically significant (albeit weaker) difference<br />

between the CT and PCA muscles in terms of the frequency<br />

of dye diffusion (p < 0.0013).<br />

Table 2 shows the results of the comparison between<br />

the genders in terms of the frequency of dye diffusion.<br />

Since there was dye diffusion to the LCA muscle in all<br />

larynges, it was impossible to perform a statistical analysis<br />

of this variable for the LCA muscle. We found that neither<br />

the width nor the height of the TA muscle had a significant<br />

influence on aniline blue diffusion to the LCA, CT, and PCA<br />

muscles (Table 3).<br />

DISCUSSION<br />

Botulinum toxin injections into the TA muscle have<br />

been widely used in the treatment of spasmodic dysphonia.<br />

Botulinum toxin is produced by the anaerobic bacterium<br />

Clostridium botulinum, and botulinum toxin type A is the<br />

Table 2. Comparison between the genders in terms of aniline<br />

blue diffusion to the lateral cricoarytenoid, cricothyroid, and<br />

posterior cricoarytenoid muscles.<br />

Gender<br />

Dye Diffusion Male Female<br />

n (%) n (%)<br />

Diffusion to the LCA muscle †<br />

Yes 14 (100%) 4 (100%)<br />

No 0 (0%) 0 (0%)<br />

Diffusion to the CT muscle*<br />

Yes 8 (57.14%) 3 (75%)<br />

No 6 (42.86%) 1 (25%)<br />

Diffusion to the PCA muscle*<br />

Yes 2 (14.29%) 0 (0%)<br />

No 12 (85.71%) 4 (100%)<br />

Note: LCA, lateral cricoarytenoid; CT, cricothyroid; PCA,<br />

posterior cricoarytenoid<br />

*p = 0.622 for the CT muscle; p = 1.000 for the PCA muscle.<br />

†<br />

Since there was dye diffusion to the LCA muscle in all larynges, it<br />

was impossible to perform a statistical analysis for this muscle.<br />

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Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy for botulinum toxin injection: an experimental study in cadaver larynges.<br />

Alonso et al.<br />

Table 3. Influence of the width and height of the thyroarytenoid<br />

muscle on aniline blue diffusion to the lateral cricoarytenoid,<br />

cricothyroid, and posterior cricoarytenoid muscles.<br />

Dye Diffusion TA Width (cm) TA Height (cm)<br />

Mean p Mean p<br />

Diffusion to the LCA muscle<br />

Yes 0.55 0.4535 0.51 0.8327<br />

No 0.51 0.52<br />

Diffusion to the CT muscle<br />

Yes 0.51 0.6272 0.53 0.7402<br />

No 0.50 0.52<br />

Diffusion to the PCA muscle<br />

Yes 0.51 0.8136 0.53 0.3110<br />

No 0.53 0.47<br />

Note: TA, thyroarytenoid; LCA, lateral cricoarytenoid; CT,<br />

cricothyroid; PCA, posterior cricoarytenoid.<br />

the LCA muscles studied showed aniline blue staining. The<br />

LCA muscle is an important adductor muscle that positions<br />

the vocal processes along the midline through the rotation<br />

of the arytenoids. Therefore, LCA muscle involvement can<br />

lead to inadequate closure of the posterior third of the<br />

glottis, with incorrect positioning of the vocal process,<br />

leading to breathiness and aspiration.<br />

Aniline blue diffusion to the CT muscle was observed<br />

in 42.86% of the cases (Figure 4). It is known that CT<br />

muscle involvement can lead to decreased fundamental<br />

frequency of phonation and, when accompanied by LCA<br />

and TA muscle paralysis, can decrease vocal intensity. The<br />

PCA muscle is the only abductor muscle of the larynx. In<br />

the present study, aniline blue diffusion to the PCA muscle<br />

was observed in 8.57% of the cases studied (Figure 5).<br />

form that is used therapeutically. Botulinum toxin blocks<br />

presynaptic acetylcholine release at the neuromuscular<br />

junction, causing partial and reversible chemodenervation<br />

leading to muscle weakness, thereby reducing spasticity in<br />

patients with dystonia (4).<br />

The most serious complication of botulinum toxin<br />

injection in the treatment of dystonia has been reported to<br />

be paralysis of muscles other than the target muscle, which<br />

is most likely attributable to toxin diffusion from the injected<br />

muscle to adjacent muscles. Various authors (14-16) have<br />

reported botulinum toxin diffusion to pharyngeal and laryngeal<br />

muscles after injection of the toxin into the<br />

sternocleidomastoid muscle for the treatment of spasmodic<br />

torticollis. According to these authors, the complications<br />

were dysphagia, hoarseness, and vocal fold paralysis. Ludlow<br />

et al (9) noted that, after injection of botulinum toxin into the<br />

TA muscle, there was transient dysphagia for liquids,<br />

breathiness, transient dyspnea, and aspiration, all of which<br />

were attributed to diffusion of the toxin to adjacent muscles.<br />

Figure 3. Lateral cricoarytenoid muscles showing aniline blue<br />

staining.<br />

The use of excised larynges allows the larynx to be<br />

cut into several sections after dye injection. However, an<br />

excised larynx has the disadvantage of being a devitalized<br />

tissue, which is immobilized (not subject to the motion that<br />

accompanies breathing, swallowing, and phonation) and<br />

without blood circulation or lymph flow. Therefore, it does<br />

not accurately reflect normal laryngeal function. In the<br />

excised larynges used in the current study, dye diffusion<br />

occurred passively, i.e., without the aid of circulation or<br />

active motion, and was probably due to hydrostatic pressure<br />

on the fluid-filled compartment of the muscle.<br />

In the present study, the LCA muscle, which is the<br />

muscle that is closest to the TA muscle, was the most<br />

affected by diffusion (Figure 3). We found that 94.29% of<br />

Figure 4. Cricothyroid muscle stained on the left.<br />

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Alonso et al.<br />

CONCLUSION<br />

Based on the results of the present study, we can<br />

conclude that the probability of botulinum toxin diffusion<br />

to muscles that are adjacent to the TA muscle is nearly<br />

100% for the LCA muscle and nearly 50% for the CT<br />

muscle. Albeit less common, diffusion of toxin to the PCA<br />

muscle can also occur. In addition, it seems that neither<br />

gender nor laryngeal size has any significant influence on<br />

the pattern of diffusion.<br />

REFERENCES<br />

1. Behlau MS, Pontes PAL, Dedo HH. Spastic Dysphonia of<br />

the focal laryngeal dystonia: the evolution of the concept<br />

of the same disease. Acta AWHO. 1991;10(2):89-95.<br />

Figure 5. Posterior cricoarytenoid muscle partially stained on<br />

both sides.<br />

Although the risk of bilateral diffusion is small, it<br />

should be taken into consideration when botulinum toxin<br />

is used in the treatment of patients with spasmodic<br />

dysphonia, particularly when the toxin is injected bilaterally,<br />

because bilateral toxin diffusion can lead to a clinical<br />

situation that is similar to bilateral adductor vocal fold<br />

paralysis. Although it is a rare complication, bilateral paralysis<br />

following botulinum toxin injection has been reported in<br />

patients with adductor spasmodic dysphonia (<strong>17</strong>).<br />

Therefore, different degrees of muscle paralysis and paresis<br />

(in isolation or in combination) can explain the different<br />

laryngeal configurations observed in such patients.<br />

Another parameter analyzed in our study was the<br />

influence of gender on aniline blue diffusion to noninjected<br />

muscles. This was tested for each muscle<br />

individually. For the LCA muscle, it was impossible to<br />

analyze this parameter because all of the larynges studied<br />

showed dye diffusion to the LCA muscle, with the exception<br />

of 2 hemi-larynges. With regards to the CT and PCA<br />

muscles, we found that gender had no statistically significant<br />

influence on dye diffusion to these muscles. Other authors<br />

have also found that there are no significant gender-related<br />

differences in diffusion (18).<br />

In addition, we tested whether the height and width<br />

of the TA muscle influenced dye diffusion to non-injected<br />

muscles. We found that neither the height nor the width of<br />

the TA muscle had any influence on dye diffusion to any<br />

of the adjacent muscles studied.<br />

2. Tsuji DH, Sennes LU, Pinho, SMR, Barbosa E. Technical<br />

application of botulinum toxin through the flexible<br />

endoscopel. In: Third Brazilian Congress from Laryngology<br />

and Voice, Annals. 1995; P 1.<br />

3. Blitzer A, Brin MF, Fahn S, et al. Botulinum toxin (BOTOX)<br />

for the treatment of “Spastic Dysphonia”as part of a trial of<br />

toxin injections for the treatment of other cranial dystonias.<br />

Laryngoscope. 1986; 96:1300-1.<br />

4. Evans CM, Williams RS, Shone, CC. Botulinum type B. Its<br />

purification, radioiodination and interaction with rat-brain<br />

synaptosomal membranes. Eur. J. Biochem.1986;154:409-<br />

16.<br />

5. Tsuji DH, Sennes LU, Imamura R, Koishi HV. Technical<br />

Injection of Botulinum Toxin by flexible endoscopel. Arq<br />

Fund Otorrinolaringol. 2001;5(3):137-43.<br />

6. Borodic GE, Joseph M, Fay L, Cozzolino D, Ferrante RJ.<br />

Botulinum A toxin for the treatment of spasmodic torticollis.<br />

Dysphagia and regional toxin spread. Otolaryngol. Head<br />

Neck Surg. 1990;12:392-8.<br />

7. Shaari CM, Ge<strong>org</strong>e E, Wu BL, Biller HF, Sanders I.<br />

Quantifying the spread of botulinum toxin through muscle<br />

fascia. Laryngoscope. 1991;101:960-4.<br />

8. Ludlow CL, Naunton RF, Sedory SE. Effects of Botulinum<br />

Toxin Injections on Speech in Adductor Spasmodic<br />

Dysphonia. Neurology. 1988;38:1220-5.<br />

9. Ludlow CL, Bagley JA, Yin SG. A comparison of<br />

different injection techniques in the treatment of<br />

spasmodic dysphonia with botulinum toxin. J. Voice.<br />

1992;6:380-6.<br />

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Diffusion of aniline blue injected into the thyroarytenoid muscle as a proxy for botulinum toxin injection: an experimental study in cadaver larynges.<br />

Alonso et al.<br />

10. Brin MF, Blitzer A, Fahn S et al. Adductor Laryngeal<br />

Dystonia (Spastic Dysphonia): Treatment with Local<br />

Injections of Botulinum Toxin (BOTOX). Mov. Disord.<br />

1989;4:287-96.<br />

11. Scott AB. Botulinum toxin injection of eye muscles to<br />

correct strabismus. Trans. Am. Ophthalmol Soc. 1981;79:734-<br />

70.<br />

12. Elston JS, Russel RW. Effects of Treatment with Botulinum<br />

Toxin on Neurogenic Blepharospasm. Br. Med. J.<br />

1985;290:1857-9.<br />

13. Hallan RI, Melling, J, Womack NR. Treatment of Anismus<br />

in Intractable Constipation with Botulinum A. Toxin. Lancet.<br />

1988;2:714-7.<br />

14. Stell R, Thompson PD, Marsden CD. Botulinum Toxin<br />

in Spasmodic Torticollis. J. Neurol. Neurosurg. Psychiatry.<br />

1988;51:920-3.<br />

15. Koay CE, Alun-Jones T. Pharyngeal Paralysis Due to<br />

Botulinum Toxin Injection. J Laryngol Otol. 1989;103:698-9.<br />

16. Liu TC, Irish, JC, Adams SG, Durkin LC, Hunt EJ.<br />

Prospective Study of Pacients’Subjective Responses to<br />

Botulinum Toxin Injection for Spasmodic Dysphonia. J<br />

Otolaryngol. 1996;25(2):66-74.<br />

<strong>17</strong>. Venkatesan NN, Johns MM, Hapner ER, DelGaudio JM.<br />

Abductor paralysis after botox injection for<br />

adductorspasmodic dysphonia. Laryngoscope. 2010<br />

Jun;120(6):1<strong>17</strong>7-80.<br />

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Original Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):321-328.<br />

DOI: 10.7162/S1809-977720130003000013<br />

Correlation of cephalometric and anthropometric measures with obstructive<br />

sleep apnea severity<br />

Paulo de Tarso M B<strong>org</strong>es 1 , Edson Santos Ferreira Filho 2 ,Telma Maria Evangelista de Araujo 3 , Jose Machado Moita Neto 4 ,<br />

Nubia Evangelista de Sa B<strong>org</strong>es 5 , Baltasar Melo Neto 6 , Viriato Campelo 7 , J<strong>org</strong>e Rizzato Paschoal 8 , Li M Li 9 .<br />

1) Master’s degree completed. Doctorate in progress. Adjunct Professor of Otolaryngology (Federal University of Piaui).<br />

2) Physician (Federal University of Piaui).<br />

3) Doctoral degree completed. Adjunct Professor of Nursing (Federal University of Piaui).<br />

4) Doctoral degree completed. Associate Professor of Chemistry (Federal University of Piaui).<br />

5) Phonoaudiologist (Paulo B<strong>org</strong>es Clinic).<br />

6) Physician (Federal University of Piaui).<br />

7) Doctoral degree completed. Associate Professor (Department of Parasitology and Microbiology, Federal University of Piaui).<br />

8) Doctoral degree completed. Associate Professor (Campinas State University (UNICAMP) School of Medicine).<br />

9) Doctoral degree completed. Full Professor (Campinas State University (UNICAMP) School of Medicine).<br />

Institution: Federal University of Piaui.<br />

Teresina / PI – Brazil.<br />

Mailing address: Paulo de Tarso Moura B<strong>org</strong>es - Av. Elias João Tajra, 1260 - Apto. 300 - Bairro: Jóquei Clube - Teresina / PI - Brazil - Zip Code: 64049-300 - Telephone:<br />

(+55 86) 3230-9797 / 3232-4306; E-mail: ptb<strong>org</strong>es@gmail.com<br />

Article received on March 7 th , 2013. Article accepted on April 7 th , 2013.<br />

SUMMARY<br />

Introduction: Patients with obstructive sleep apnea-hypopnea syndrome (OSAHS) often have associated changes in craniofacial<br />

morphology and distribution of body fat, either alone or in combination.<br />

Aim: To correlate cephalometric and anthropometric measures with OSAHS severity by using the apnea-hypopnea index (AHI).<br />

Method: A retrospective cephalometry study of 93 patients with OSAHS was conducted from July 2010 to July 2012. The<br />

following measurements were evaluated: body mass index (BMI), neck circumference (NC), waist circumference (WC), hip<br />

circumference (HC), the angles formed by the cranial base and the maxilla (SNA) and the mandible (SNB), the difference between<br />

SNA and SNB (ANB), the distance from the mandibular plane to the hyoid bone (MP-H), the space between the base of the<br />

tongue and the posterior pharyngeal wall (PAS), and the distance between the posterior nasal spine and the tip of the uvula<br />

(PNS-P). Means, standard deviations, and Pearson’s correlation coefficients were calculated and analyzed.<br />

Results: AHI correlated significantly with BMI (r = 0.207, p = 0.047), NC (r = 0.365, p = 0.000), WC (r = 0.337, p = 0.001),<br />

PNS-P (r = 0.282, p = 0.006), and MP-H (r = 0.235, p = 0.023).<br />

Conclusion: Anthropometric measurements (BMI, NC, and WC) and cephalometric measurements (MP-H and PNS-P) can be<br />

used as predictors of OSAHS severity.<br />

Keywords: Cephalometry; Sleep Apnea, Obstructive; Anthropometry; Body Mass Index; Abdominal Circumference; Waist<br />

Circumference.<br />

INTRODUCTION<br />

Obstructive sleep apnea-hypopnea syndrome<br />

(OSAHS) is a disorder characterized by recurrent episodes<br />

of partial or total upper airway obstruction during sleep.<br />

The apnea-hypopnea index (AHI) refers to the number of<br />

episodes of apnea and hypopnea that occur per hour of<br />

sleep (1).<br />

OSAHS affects 4–7% of the general adult population<br />

(2). Owing to its current prevalence, it is considered a<br />

major public health concern, which can manifest serious<br />

physical and social consequences if not managed properly<br />

(2,3). This disorder mainly affects middle-aged economically<br />

active patients, resulting in high costs and lost workdays<br />

(4). Medical costs can be significantly reduced when<br />

effective diagnosis and treatment are performed early (5).<br />

The diagnosis of OSAHS is based on a combination of the<br />

laboratory findings of apnea and hypopnea with clinical<br />

symptoms (6).<br />

Radiographic imaging of the upper airways allows<br />

for the study of bone and soft tissue anatomy, in addition<br />

to the determination of the site of obstruction and the<br />

choice of appropriate treatment for OSAHS patients (7).<br />

Cephalometry has been used since 1983 and was initially<br />

applied in patients with sleep-related breathing disorders.<br />

It consists of cephalometric tracings obtained by<br />

teleradiography of the facial profile. The aim of the test is<br />

to study the facial, maxillary, and mandibular skeleton and<br />

any relationships with the soft tissues that may cause<br />

pharyngeal obstruction (8). Cephalometry is an easy, lowcost,<br />

non-invasive, and widely available modality in the<br />

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B<strong>org</strong>es et al.<br />

majority of hospitals. Radiation use is minimal, readily<br />

accessible to clinicians, and not uncomfortable for the<br />

patient. It has proved to be useful for the evaluation of the<br />

upper airways and bone structure in normal subjects and<br />

OSAHS patients (9-11).<br />

A complete cephalometric analysis should be<br />

performed in patients with OSAHS to identify associated<br />

maxillofacial abnormalities (12). From this perspective, it<br />

has been used in several sleep disorder centers, with the<br />

purpose of diagnosing the site of obstruction in patients<br />

with obstructive sleep apnea. In some centers, this test is<br />

part of a service protocol that is important for the decision<br />

as to which type of surgery should be performed. Therefore,<br />

it is recommended in all patients with OSAHS undergoing<br />

surgery (8,9,12-15). It is also used with mandibular<br />

advancement devices to assess therapeutic efficacy in<br />

patients with mild to moderate OSAHS (15,16).<br />

Obesity, in particular the presence of visceral fat, is<br />

considered a predictive factor for OSAHS (<strong>17</strong>). Several<br />

studies have been performed using anthropometric<br />

measurements of obesity in patients with OSAHS, e.g.,<br />

body mass index (BMI), neck circumference (NC), waist<br />

circumference (WC), and hip circumference (HC) (18-21).<br />

These measurements may be used both to assess the need<br />

for patient referral to polysomnographic evaluation and to<br />

anticipate treatment in high-risk patients, since this disorder<br />

may cause severe consequences in untreated patients<br />

(2,21). Polysomnography is not an accessible test for most<br />

individuals. It is expensive and easier, more affordable tests<br />

with less technological density, such as cephalometry (9-<br />

11), are required (<strong>17</strong>,19,22,23) to screen for OSAHS.<br />

Anthropometric measurements are easily obtained and<br />

fundamental for the preparatory study of a patient with<br />

suspected OSAHS. Therefore, the number of patients<br />

referred for polysomnography could be greatly reduced,<br />

lowering healthcare expenses, with simple measurement<br />

of cervical, waist, and hip circumferences. The use of these<br />

measurements in patients with snoring and those clinically<br />

suspected of having OSAHS would thus prioritize<br />

complementary polysomnography testing in patients with<br />

higher suspicion of disease (24).<br />

The aim of this study was to correlate cephalometric<br />

and anthropometric measurements with the AHI, in order<br />

to assess if these measurements can be used as predictors<br />

of OSAHS severity.<br />

METHOD<br />

Data obtained from the medical charts of 93 male<br />

and female patients with OSAHS, ranging in age from 19 to<br />

80 years, were studied. These patients had been examined<br />

from July 2010 to July 2012 in a specialist private clinic in<br />

Teresina. The medical charts of patients who had undergone<br />

previous surgical treatment for OSAHS, including use of<br />

continuous positive airway pressure devices or intraoral<br />

devices, in addition to patients with craniofacial deformities<br />

and upper airway tumors, were excluded from the study.<br />

Overnight polysomnography was analyzed by a<br />

single professional, who specialized in sleep disorders.<br />

Patients were considered to have OSAHS if, in addition to<br />

clinical complaints, they had an AHI > 5 upon overnight<br />

polysomnography testing (1). Severity was measured by<br />

the AHI. Cephalometry was also performed by a single<br />

radiologist, who specialized in orthodontic radiographs.<br />

Each examiner was blinded to the test results obtained by<br />

the other examiner.<br />

The following cephalometric traces were considered,<br />

since they are the most commonly used cephalometric<br />

measures: retroglossal posterior airway space (PAS), defined<br />

as the space between the base of the tongue and the<br />

posterior pharyngeal wall; the distance between the mandibular<br />

plane and the hyoid bone (MP-H); the SNA angle,<br />

formed by the junction between the sellar point (S,<br />

midpoint of the sella turcica), nasion (N, junction between<br />

the frontal and nasal bones), and point A (deepest concavity<br />

on the anterior profile of the maxilla); the SNB angle,<br />

formed by the junction between the sellar point (S), nasion<br />

(N), and point B (deepest concavity on the anterior profile<br />

of the mandibular symphysis); the distance between the<br />

posterior nasal spine and the tip of the soft palate (PNS-P);<br />

and the difference between SNA and SNB (ANB) (Figure 1)<br />

(8,9,12,15).<br />

Concerning anthropometric measurements, BMI,<br />

NC, WC, and HC were evaluated. BMI was calculated as the<br />

weight of the subject (in kilograms) divided by their height<br />

(in meters) squared. Obesity is defined as a BMI > 30 kg/<br />

m 2 (25). NC was measured at the level of the crycothyroid<br />

cartilage. WC was measured between the last rib and the<br />

iliac crest and HC was measured as the maximum<br />

circumference at the level of the border of the trochanter.<br />

Anthropometric measurements were based on the World<br />

Health Organization manual (26).<br />

Data was entered into the Statistical Package for the<br />

Social Sciences (SPSS) version 16.0 program for calculation<br />

of simple descriptive statistics, e.g., percentage distribution,<br />

mean, and standard deviation. A normality test (Kolmogorov-<br />

Smirnov) was applied and the adequate statistical test was<br />

chosen for each type of variable (parametric or<br />

nonparametric). Pearson’s correlation coefficients were<br />

examined between AHI and the following variables: age,<br />

BMI, NC, WC, HC, and cephalometric measurements (SNA,<br />

SNB, ANB, MP-H, PAS, and PNS-P). Sex differences were<br />

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Table 1. Sample distribution according to gender and obesity.<br />

n % BMI, kg/m 2<br />

(mean ± SD)<br />

Gender Male 54 58.06 27.88 ± 3.30<br />

Female 39 41.94 27.41 ± 4.50<br />

Total 93 100.00 27.68 ± 3.83<br />

Obesity Nonobese 69 74.19 25.90 ± 2.29<br />

Obese 24 25.81 32.81 ± 2.50<br />

Total 93 100.00 27.68 ± 3.83<br />

Note: BMI = body mass index.<br />

Figure 1. Description of cephalometric parameters (Source:<br />

the author).<br />

compared by the Student’s t test. Statistical significance<br />

was set at p < 0.05.<br />

The project was approved by the Research Ethics<br />

Committee of the Universidade Federal do Piauí, under<br />

CAAE number 0047.0.045.000-10.<br />

RESULTS<br />

Of the 93 patients studied, 54 (58.1%) were male.<br />

The mean BMI was 27.68 ± 3.83 kg/m 2 (women, 27.41 ±<br />

4.50 kg/m 2 ; men, 27.88 ± 3.30 kg/m 2 ). There were 69<br />

(74.19%) nonobese (BMI, 25.90 ± 2.29 kg/m 2 ) and 24<br />

(25.81%) obese (BMI, 32.81 ± 2.50 kg/m 2 ) patients<br />

(Table 1).<br />

The mean age of the patients was 46.70 ± 15.46<br />

years (range, 19 to 80 years). Table 2 describes the sample<br />

characteristics and variables studied, including their variations<br />

and means.<br />

Table 3 shows an analysis of the Pearson correlation<br />

coefficients used to evaluate the relationship between<br />

AHI, age, and anthropometric/cephalometric<br />

measurements. Analyzing the relationship between age<br />

and OSAHS severity by AHI showed a positive correlation<br />

between these 2 data series, indicating that OSAHS was<br />

more severe in older patients. Similarly, there was also a<br />

Table 2. Characteristics of the sample including age,<br />

anthropometric parameters, cephalometric parameters, and<br />

AHI (n = 93).<br />

Variable Minimum Maximum Mean Standard<br />

deviation<br />

Age, years 19.0 80.0 46.70 15.46<br />

BMI, kg/m 2 19.72 40.18 27.68 3.83<br />

NC, cm 30.0 47.0 38.56 3.92<br />

WC, cm 73.0 125.0 97.59 10.10<br />

HC, cm 87.0 131.0 104.09 7.39<br />

SNAº 72.0 92.0 82.77 4.08<br />

SNBº 69.0 92.0 80.96 4.41<br />

ANBº -13.0 10.0 1.82 3.90<br />

MP-H, mm 4.0 45.0 19.21 8.22<br />

PAS, mm 3.0 20.0 10.04 3.80<br />

PNS-P, mm 20.0 52.0 39.84 5.37<br />

AHI, events/h 5.00 83.40 34.67 <strong>17</strong>.41<br />

Note: AHI = apnea-hypopnea index; BMI = body mass index;<br />

NC = neck circumference; WC = waist circumference;<br />

HC = hip circumference; SNA = angle formed by the junction<br />

of the sella (S), nasion (N), and point A; SNB = angle formed<br />

by the junction of the sella (S), nasion (N), and point B;<br />

ANB = difference between SNA and SNB; MP-H = distance<br />

between the mandibular plane and the hyoid bone;<br />

PAS = space between the base of the tongue and the posterior<br />

pharyngeal wall; PNS-P = distance between the posterior<br />

nasal spine and the tip of the soft palate.<br />

positive correlation between BMI and AHI, which allowed<br />

us to infer that the higher the BMI, the greater the AHI.<br />

Regarding body circumference measurements, both NC<br />

and WC showed a statistically significant positive correlation<br />

with OSAHS severity. Concerning the relationship between<br />

cephalometric data and AHI, a statistically significant positive<br />

correlation for MP-H and PNS-P was observed (Table 3).<br />

Comparing anthropometric parameters between<br />

men and women indicated a statistically significant difference<br />

for NC and WC, but not for HC. Regarding cephalometry,<br />

MP-H and PNS-P were significantly different between men<br />

and women (Table 4).<br />

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Table 3. Correlation of age and anthropometric/cephalometric data with AHI (n = 93).<br />

Age BMI NC WC HC SNA SNB ANB MP-H PAS PNS-P<br />

Pearson correlation 0.241 0.207 0.365 0.337 0.201 -0.044 0.031 -0.081 0.235 -0.102 0.282<br />

p-value (two-tailed) 0.020 0.047 0.000 0.001 0.053 0.676 0.769 0.441 0.023 0.329 0.006<br />

Note: AHI = apnea-hypopnea index; BMI = body mass index; NC = neck circumference; WC = waist circumference; HC = hip<br />

circumference; SNA = angle formed by the junction of the sella (S), nasion (N), and point A; SNB = angle formed by the junction<br />

of the sella (S), nasion (N), and point B; ANB = difference between SNA and SNB; MP-H = distance between the mandibular<br />

plane and the hyoid bone; PAS = space between the base of the tongue and the posterior pharyngeal wall; PNS-P = distance<br />

between the posterior nasal spine and the tip of the soft palate<br />

Tables 5 and 6 show an analysis of the Pearson<br />

correlation coefficients used to assess the relationship<br />

between AHI and age as well as anthropometric/<br />

cephalometric measurements for males and females.<br />

For anthropometric measurements, AHI showed a<br />

statistically significant correlation with age, BMI, NC, and<br />

WC in males. Only NC showed a significant correlation in<br />

females. In both genders, the most significant correlation<br />

was with NC (Table 5). Regarding cephalometric<br />

measurements, the only correlation found was that of AHI<br />

with PNS-P measurement in males (Table 6).<br />

DISCUSSION<br />

Obesity may increase susceptibility to OSAHS by<br />

causing fat deposition in the upper airway tissues, narrowing<br />

the nasopharyngeal caliber and/or leading to hypoventilation<br />

in association with reduced wall complacency (27).<br />

Assessment of craniofacial morphology in OSAHS patients<br />

not only aids specialists concerned with recognizing<br />

morphologic changes induced by altered sleep patterns,<br />

but also provides the patient with adequate treatment<br />

(11).<br />

There is a vast amount of scientific literature on<br />

cephalometric and anthropometric measures, which compares<br />

control groups and snorers to OSAHS patients and<br />

aims at using these measurements as predictors of this<br />

condition (7,9-11,13,15,18,20,22,28-32). Some studies have<br />

been performed to assess appropriate treatments, surgical<br />

treatment plans, and the indications of intraoral devices<br />

(11,16,27,33-40), while others were performed to evaluate<br />

the relationship between these measurements and OSAHS<br />

severity (<strong>17</strong>-20,22-24,32,40-45).<br />

In the current study, there was a significant correlation<br />

between age and AHI (Table 3), indicating that OSAHS is<br />

more severe in older patients. This is in agreement with the<br />

majority of previous studies (<strong>17</strong>,22-24,45,46), with the<br />

exception of those by Mayer et al42 and Schellenberg,<br />

Maisline, and Schwab (37).<br />

Table 4. Comparison between males and females for<br />

anthropometric/cephalometric data and AHI.<br />

Females (n = 39) Males (n = 54) p-value<br />

Age, years 49.77 ± 14.32 44.48 ± 15.99 p > 0.05<br />

BMI, kg/m 2 27.41 ± 4.50 27.88 ± 3.30 p > 0.05<br />

NC, cm 35.31 ± 2.27 40.91 ± 3.09<br />

**<br />

p < 0.01<br />

WC, cm 94.41 ± 10.79 99.89 ± 8.99<br />

**<br />

p < 0.01<br />

HC, cm 102.56 ± 8.65 105.19 ± 6.18 p > 0.05<br />

SNAº 82.74 ± 3.38 82.80 ± 4.56 p > 0.05<br />

SNBº 80.41 ± 4.35 81.35 ± 4.44 p > 0.05<br />

ANBº 2.33 ± 3.98 1.44 ± 3.83 p > 0.05<br />

MP-H, mm 15.04 ± 6.62 22.22 ± 8.00<br />

**<br />

p < 0.01<br />

PAS, mm 9.28 ± 3.44 10.59 ± 3.98 p > 0.05<br />

PNS-P, mm 37.82 ± 5.10 41.30 ± 5.12<br />

**<br />

p < 0.01<br />

AHI, events/h 31.02 ± 15.20 37.30 ± 18.53 p > 0.05<br />

Note: AHI = apnea-hypopnea index; BMI = body mass index;<br />

NC = neck circumference; WC = waist circumference;<br />

HC = hip circumference; SNA = angle formed by the junction<br />

of the sella (S), nasion (N), and point A; SNB = angle formed<br />

by the junction of the sella (S), nasion (N), and point B;<br />

ANB = difference between SNA and SNB; MP-H = distance<br />

between the mandibular plane and the hyoid bone;<br />

PAS = space between the base of the tongue and the posterior<br />

pharyngeal wall; PNS-P = distance between the posterior nasal<br />

spine and the tip of the soft palate<br />

It was observed that BMI was correlated with AHI,<br />

allowing us to infer that more severe OSAHS occurs in<br />

subjects with a higher BMI, which is concordant with most<br />

published studies (<strong>17</strong>,19,21-23,31,32,37,39,42,45-47).<br />

Nevertheless, this fact was not observed by Yucel et al (41)<br />

and Martinez-Rivera et al (24).<br />

NC is a simple measurement that is easily performed.<br />

Findings in the present study point to a positive correlation<br />

between NC and AHI, which is in agreement with other<br />

studies (19,20,22,24,31,32,37-39,41,46). A correlation with<br />

BMI was also observed previously (<strong>17</strong>-19,23,32,44),<br />

suggesting that obesity affects patients with OSAHS through<br />

fat deposition in the neck (20,23,30). In contrast to the<br />

majority of studies, Ogretmenoglu et al (<strong>17</strong>) stated that NC<br />

was more weakly correlated with AHI than BMI.<br />

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Table 5. Pearson correlation of AHI with age and anthropometric data for males and<br />

females.<br />

Age BMI NC WC HC<br />

M Pearson correlation 0.358 0.273 0.349 0.395 0.156<br />

p-value (two-tailed) 0.008 ** 0.046 * 0.010 ** 0.003 ** 0.259<br />

n 54 54 54 54 54<br />

F Pearson correlation 0.131 0.123 0.329 0.192 0.212<br />

p-value (two-tailed) 0.426 0.457 0.041 * 0.242 0.195<br />

n 39 39 39 39 39<br />

Note: AHI = apnea-hypopnea index; M = males; F = females; BMI = body mass index;<br />

NC = neck circumference; WC = waist circumference; HC = hip circumference;<br />

** p < 0.01; * p < 0.05.<br />

Table 6. Pearson correlation between AHI and cephalometric data for males and females.<br />

SNA SNB ANB MP-H PAS PNS-P<br />

M Pearson correlation -0.075 0.031 -0.125 0.229 -0.212 0.305<br />

p-value (two-tailed) 0.592 0.822 0.368 0.096 0.124 0.025 *<br />

n 54 54 54 54 54 54<br />

F Pearson correlation 0.020 -0.021 0.040 0.073 0.010 0.135<br />

p-value (two-tailed) 0.904 0.898 0.809 0.660 0.951 0.412<br />

n 39 39 39 39 39 39<br />

Note: AHI = apnea-hypopnea index; M = males; F = females; SNA = angle formed by the junction<br />

of the sella (S), nasion (N), and point A; SNB = angle formed by the junction of the sella (S),<br />

nasion (N), and point B; ANB = difference between SNA and SNB; MP-H = distance between<br />

the mandibular plane and the hyoid bone; PAS = space between the base of the tongue and<br />

the posterior pharyngeal wall; PNS-P = distance between the posterior nasal spine and the tip<br />

of the soft palate; * p < 0.05.<br />

WC also showed a positive correlation with AHI, in<br />

agreement with other studies (19,23,24,32,38,39,46).This<br />

correlation was weaker than that observed with NC,<br />

supporting the existing literature (19,23,39). The present<br />

study is also consistent with others (19,32) showing that<br />

the correlation of AHI with WC is greater than that with<br />

BMI. However, Davidson and Patel (32) observed that WC<br />

was more predictive than BMI as well as NC.<br />

Analyzing the relationship between cephalometric<br />

data and AHI showed a statistically significant correlation<br />

for MP-H and PNS-P, indicating an increased palatal length<br />

and an increased distance between the hyoid bone and the<br />

mandibular plane, which was consistent with other studies<br />

(29,42,43,46,48). According to Yucel et al (41), only MP-<br />

H measurement is related to AHI. The position of the hyoid<br />

bone, which has an impact on the shape and position of the<br />

tongue, affects hypopharyngeal airway patency (41).<br />

A study by Bharadwaj, Ravikumar, and Krishnaswamy<br />

conducted in India compared 10 OSAHS patients to a<br />

control group of 10 healthy subjects (mean age, 34.9<br />

years). These authors concluded that upright cephalometry<br />

demonstrated mandibular retrognathism, increased ANB<br />

angle, increased PNS-P length and increased soft palate<br />

thickness, increased tongue length, and decreased PAS in<br />

the group of OSAHS patients when compared to the<br />

control group (11). Other studies have shown that PNS-P<br />

and MP-H measurements are increased, while PAS is<br />

decreased, in patients with OSAHS, and these measurements<br />

were considered predictive of OSAHS (7,9,15). PAS<br />

measurement was correlated with severity of OSAHS<br />

assessed using the AHI (7,9). A study conducted in<br />

Strasbourg (France) in 1990, comparing 43 OSAHS patients<br />

to a control group of 40 asymptomatic individuals, showed<br />

that PNS-P measurement was increased in patients with<br />

OSAHS, while SNA, SNB, and ANB measurements did not<br />

demonstrate any alterations in either group (28). Other<br />

studies have shown increased MP-H measurement and<br />

decreased PAS measurement in comparisons between<br />

OSAHS groups and control groups (13,40). However,<br />

Mayer et al (42) found no correlation between PAS<br />

measurement and AHI.<br />

In a comparison between male and female patients<br />

in the present study, BMI and HC were similar. NC and WC<br />

were significantly higher in males than in females. These<br />

results are partially in agreement with findings by Millman<br />

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B<strong>org</strong>es et al.<br />

et al (49), who described a higher NC for males, but no<br />

difference between the genders for BMI and WC. Another<br />

study demonstrated a significantly increased BMI in females<br />

(41). This fact reflects well-known characteristics concerning<br />

body fat distribution in men and women (49).<br />

In the current study, NC was more closely correlated<br />

with AHI than WC and BMI, both in the overall population<br />

and in male and female subjects, indicating that NC is the<br />

best anthropometric measurement for prediction of OSAHS<br />

severity, especially in males (18,19,23,39,41,44).<br />

Comparing cephalometry between male and female<br />

subjects showed a statistically significant difference in<br />

MP-H and PNS-P measurements, which were higher in<br />

male subjects. PNS-P measurement was correlated with<br />

AHI in males, while there were no significant correlations<br />

in females.<br />

In conclusion, the results of this study show a<br />

correlation of anthropometric (BMI, NC, and WC) and<br />

cephalometric (MP-H and PNS-P) measurements with the<br />

AHI, reinforcing their preparatory role and use as predictors<br />

of OSAHS.<br />

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Review Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):329-339.<br />

DOI: 10.7162/S1809-977720130003000014<br />

Use of surface electromyography in phonation studies: an integrative<br />

review<br />

Patricia Maria Mendes Balata 1 , Hilton Justino da Silva 2 , Kyvia Juliana Rocha de Moraes 3 , Leandro de Araújo Pernambuco 4 ,<br />

Sílvia Regina Arruda de Moraes 5 .<br />

1) Doctor in Neuropsychiatry and Behaviour Science of Adolescent Health; Speech and Language Pathologist; Voice Specialist ; (Speech and Language Pathologist of<br />

the Hospital dos Servidores do Estado de Pernambuco).<br />

2) Doctor of Nutrition; Speech and Language Pathologist; Master of Anatomy; Specialist in oral Motricity (Teacher in the Speech and Language Pathology Department<br />

of the Universidade Federal de Pernambuco).<br />

3) Master of Pathology; Physiotherapist (Teacher in the Physiotherapy Department of the Faculdade Estacio do Recife).<br />

4) Master of Pathology; Speech and Language Pathologist; Specialist in oral Motricity (Teacher in the Speech and Language Pathology Department of the Universidade<br />

Federal do Rio Grande do Norte; Doctoral Student in Public Health).<br />

5) Doctor of Sciences; Physiotherapist (Teacher in the Anatomy Department of the Universidade Federal de Pernambuco).<br />

Institution: Universidade Federal de Pernambuco.<br />

Recife / PE – Brazil.<br />

Mailig address: Patricia Maria Mendes Balata - Avenida Domingos Ferreira, 636, sala 208 - Pina - Recife / PE – Brazil - Zip Code: 51011-010 - Telephone: (+55 81)<br />

3326-9482 - E-mail: pbalata@uol.com.br.<br />

Financial support from National Council for Scientific and Technological Development with Edictal Universal Process MCT/CNPQ 14/2009, B, process: 476412/2009.<br />

Article received on October 4 th , 2012. Article accepted on January 18 th , 2013.<br />

SUMMARY<br />

Introduction: Surface electromyography has been used to assess the extrinsic laryngeal muscles during chewing and swallowing,<br />

but there have been few studies assessing these muscles during phonation.<br />

Objective: To investigate the current state of knowledge regarding the use of surface electromyography for evaluation of the<br />

electrical activity of the extrinsic muscles of the larynx during phonation by means of an integrative review.<br />

Method: We searched for articles and other papers in the PubMed, Medline/Bireme, and Scielo databases that were published<br />

between 1980 and 2012, by using the following descriptors: surface electromyography and voice, surface electromyography<br />

and phonation, and surface electromyography and dysphonia. The articles were selectedon the basis ofinclusion and exclusion<br />

criteria.<br />

Data Synthesis: This was carried out with a cross critical matrix. We selected 27 papers,i.e., 24 articles and 3 theses. The studies<br />

differed methodologically with regards to sample size and investigation techniques, making it difficult to compare them, but<br />

showed differences in electrical activity between the studied groups (dysphonicsubjects, non-dysphonicsubjects, singers, and<br />

others).<br />

Conclusion: Electromyography has clinical applicability when technical precautions with respect to application and analysis<br />

are obeyed. However, it is necessary to adopt a universal system of assessment tasks and related measurement techniques to<br />

allow comparisons between studies.<br />

Keywords: Electromyography; Phonation; Dysphonia; Laryngeal Muscles.<br />

INTRODUCTION<br />

The increased use of surface electromyography<br />

(SEMG) in speech and language pathology, not only in<br />

the literature but also in clinical practice, makes it<br />

important to investigate the current state of knowledge<br />

regarding this technique. This is true especially in the<br />

area of orofacial motricity, where SEMG is used as a tool<br />

for assessment and treatment. SEMG is also utilized in<br />

studies on the voice. When assessing the current<br />

knowledge regarding a particular subject area, it is<br />

important to identify and document the best evidence<br />

produced by quantitative and qualitative research on the<br />

topic in question. Then, through integrative review, it is<br />

possible to inventory gaps in the current knowledge that<br />

may constitute new challenges to the scientific<br />

community.<br />

Electromyography is a technique that measures the<br />

electrical activity (EA) of several muscles in the body in<br />

order to diagnose movement disorders, and can also<br />

contribute to the assessment of prognosis in motor alterations.<br />

It has been employed in the fields of neurology, orthopedics,<br />

physical therapy, and otorhinolaryngology. There are 2<br />

types of electromyographic examinations. Insertion<br />

electromyography (EMG) evaluates the action potentials<br />

of the motor units of deeper muscles; it is invasive and<br />

characterized by the use of needle electrodes fixed to the<br />

muscles under investigation. SEMG uses electrodes attached<br />

to the dermis to capture the myoelectric signals from<br />

muscles or muscle groups closer to the skin surface.<br />

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Balata et al.<br />

In speech and language pathology, SEMG is used to<br />

aid in diagnosis and therapy, especially for the orofacial<br />

functions of chewing and swallowing. These functions<br />

have a close relationship with movement of the larynx; the<br />

main function of the larynx is protection of the upper<br />

airways during the act of swallowing. To understand the<br />

activity of the intrinsic muscles of the larynx, the field of<br />

otorhinolaryngology has used EMG mainly for investigating<br />

neurological voice disorders. However, there has been<br />

little research on the extrinsic laryngeal muscles. These<br />

muscles have their origin in extra-laryngeal structures, but<br />

modify phonation indirectly by either raising or lowering<br />

the larynx via the basic function of the suprahyoid (SH)<br />

muscles and infrahyoid (IH) muscles, respectively. The<br />

evaluation of these muscles by SEMG is a procedure<br />

considered feasible for clinical examination.<br />

The objective of this study was to investigate the<br />

current state of knowledge regarding the use of SEMG in<br />

assessing the EA of the SH and IH extrinsic muscles during<br />

phonation in order to help in understanding the complex<br />

phonatory phenomenon and its disorders, as well as to<br />

certify the use of this tool in vocal clinics.<br />

METHOD<br />

The integrative review is a method that belongs to<br />

the systematic review technique which, in turn, is focused<br />

on experimental studies that are preferably randomized<br />

with rigorous control of variables. This allows the safe<br />

development of evidence-based practice (1). As an<br />

alternative to narrative and systematic reviews, the<br />

integrative review involves a broader approach that allows<br />

the inclusion of studies which are not only experimental<br />

but also quasi-experimental or non-experimental. The<br />

process involved systematically follows pre-defined steps<br />

and allows a greater scope of theoretical and empirical<br />

sampling through a rigorous examination process.<br />

The following steps comprised the present study:<br />

(1) preparation of the guiding question; (2) literature<br />

search for the definition of descriptors; (3) selection of<br />

articles according to the criteria for inclusion and exclusion;<br />

(4) collection, data extraction, reading, and critical analysis;<br />

(5) interpretation and discussion of results; (6) knowledge<br />

synthesis and review presentation.<br />

Step 1) Preparation of the guiding question: the<br />

guiding question “what is the current state of knowledge<br />

regarding the use of SEMG in studies on the voice?” was<br />

defined from the theoretical and practical knowledge of<br />

the authors, with regards to the fact that SEMG is being<br />

increasingly used in studies on the area of speech and the<br />

voice, especially in Brazil.<br />

Step 2) Literature search for the definition of<br />

descriptors: articles were searched for on the Pubmed,<br />

Medline, and Scielo databases using the following descriptors:<br />

surface electromyography and voice, surface<br />

electromyography and phonation, and surface<br />

electromyography and dysphonia.<br />

Step 3) Selection of articles according to the criteria<br />

for inclusion and exclusion: articles and theses produced<br />

between 1980 and 2012 that complied with the criteria for<br />

inclusion and exclusion (Table 1) were included. Initially,<br />

we identified 48 articles on PubMed, 40 articles on Medline/<br />

Bireme, and 2 articles on Scielo using the descriptor surface<br />

electromyography and phonation. Due to the quantitative<br />

difference between Pubmed and Medline/Bireme, a new<br />

search with the same descriptors was performed on the<br />

Pubmed Central database. This time, 114 publications<br />

were found with the first descriptor surface<br />

electromyography and voice. With the other descriptors,<br />

the amount of publications found was lower, and all other<br />

articles had already been identified in the initial search<br />

(Figure 1).<br />

Step 4) Collection, data extraction, reading, and<br />

critical analysis: these steps resulted in the development of<br />

an analytical matrix consisting of the following: article title,<br />

authors, country of origin, year of publication, type of<br />

study, study objective, sample, method (including vocal<br />

tasks and SEMG procedures), and conclusions (Table<br />

2).These variables were considered to respond to the<br />

objective of the current review.<br />

Steps 5) and 6) Interpretation and discussion of<br />

results and knowledge synthesis and review presentation:<br />

Table 1. Inclusion and exclusion criteria for study selection<br />

Inclusion criteria<br />

Exclusion criteria<br />

Articles and theses produced Studies involving functions<br />

between 1980 to 2012 in other than phonation<br />

English, Portuguese, and<br />

Spanish<br />

Experimental studies,<br />

Studies restricted to description<br />

quasi-experimental studies, and evaluation of the technical<br />

or non-experimental studies characteristics of equipment<br />

Studies addressing the use Studies addressing the use of<br />

of SEMG as an assessment SEMG as a training tool<br />

tool fornormal and abnormal (biofeedback)<br />

human voice<br />

Studies evaluating the<br />

Studies on patients with<br />

extrinsic muscles of the alterations that prevent<br />

larynx and cervical muscles phonation<br />

during vocal tasks<br />

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Balata et al.<br />

Researches published from 1980 to 2012<br />

(PubMed/Medline, Scielo and Bireme) N=90<br />

New search in PubMed<br />

Central N=76<br />

Selected studies for integral reading by three<br />

judges<br />

N = 38<br />

Select articles for abstract analyses N=114<br />

Themes other than the objective<br />

N=52<br />

Duplicity N=66<br />

Researches selected for complete reading<br />

N=36<br />

Researches selected to<br />

integrate this review<br />

N=27<br />

Themes other than the objective N=09<br />

Idiom inadequacy N=03<br />

Other procedures N=09<br />

Figure 1. Scheme for searching and selection of works<br />

according to the inclusion and exclusion criteria.<br />

analysis and discussion of the results were performed<br />

descriptively and integratively. This was followed by the<br />

sixth step that consisted of the synthesis of knowledge and<br />

review presentation.<br />

RESULTS AND DISCUSSION<br />

The final selection of studies using the inclusion and<br />

exclusion criteria resulted in 24 articles and 3 theses being<br />

included for review; the theses consisted of a dissertation<br />

and 2 doctoral theses. Most studies included in this review<br />

originated from the United States (14 studies), followed by<br />

5 articles from Brazil, 6 articles from European countries,<br />

and 1 article from Australia. This indicates the decentralization<br />

of interest in the use of SEMG technology to aid in<br />

understanding of the phonation phenomenon (Table 2).<br />

While the use of SEMG in studies on voice began in<br />

the 1980s (2 studies), the number of studies increased in<br />

the 1990s (5studies) and from the year 2000 onwards (20<br />

studies). From 2004, publications in this field had an annual<br />

frequency. This shows the growing interest of the scientific<br />

community in contributing more quantitative data on vocal<br />

assessment, which in turn has conferred greater objectivity<br />

to the parameters of this function, with the aim of<br />

understanding the behavior of the extrinsic muscles in<br />

various situations of phonation through the use of SEMG.<br />

The studies investigated had diverse methodology,<br />

which made it difficult to compare them. The studies<br />

differed with respect to the muscle groups investigated,<br />

the tasks performed, and the sample size. There were<br />

often less than 40 subjects distributed between cases and<br />

controls (n = 25) (2-11), and some studies did not include<br />

comparison groups (n = 15) (12-25). However, controlled<br />

studies did show differences between the EA of muscles<br />

evaluated in case and control groups, and attested to the<br />

quality of SEMG as a tool for clinical evaluation of the voice.<br />

Regarding the main objectives of the articles, there<br />

was a high frequency of articles studying hyperfunctional<br />

dysphonia (n = 7) (6,8,10,11,13,14,26), probably as a result<br />

of the muscle tension present in these frameworks and the<br />

need to bring greater objectivity to the assessment of these<br />

disorders. Other types of functional dysphonia, such as<br />

nodules and glottic chinks, were the subject of interest in<br />

5 studies (4,7,9,16,27), and laryngectomized patients were<br />

subject of 3 studies (5,15,21). The muscle activity involved<br />

in singing was also a subject of interest (n = 4) (4,<strong>17</strong>,19,24).<br />

SEMG was used in the cervical and extrinsic laryngeal<br />

muscles, and the findings were related to the evaluation of<br />

various phonatory tasks such as usual phonation in the<br />

emission of vowels, connected speech and reading,<br />

simulated phonation in hyperfunction and whisper, singing<br />

voice, and the voice at rest.<br />

Concerning electrode allocation, there was no<br />

normalization in the articles selected. Electrode allocation<br />

differed as a result of the objective of each study and the lack<br />

of an institutional recommendation regarding research<br />

procedures, especially for the muscles of small caliber that<br />

make up the SH, IH, and orofacial muscles (such as the lips<br />

orbicular (LO) and masseter (MA) muscle). Scientific<br />

<strong>org</strong>anizations such as the International Society of<br />

Electrophysiology and Kinesiology (ISEK) and the group<br />

Surface EMG for the Non-Invasive Assessment of Muscles<br />

(SENIAM) integrate the basic research conducted in this area<br />

and enable exchange of data and experience with SEMG.<br />

These <strong>org</strong>anizations have established appropriate criteria on<br />

electrode allocation and methods for captured signal<br />

processing, but make no reference to the muscle groups<br />

involved in phonation such as the SH and IH muscles.<br />

The reason for election of unilateral electrode<br />

allocation in 12 studies is not clear (4,6,10,12-<br />

15,18,19,21,22,25), especially when the equipment used<br />

had channels to support bilateral investigation of muscle<br />

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Balata et al.<br />

activity. Bilateral evaluation of muscle activity may have<br />

identified possible asymmetries, although it does not<br />

always produce clear findings, as shown by the results of<br />

8 studies (5,7,9,16,20,23,24,26). Therefore, the<br />

recommendation made in the study by Stepp (28) regarding<br />

compliance with the appropriate use of bipolar electrodes,<br />

which stated that they should be used in the body of the<br />

muscle of interest and never bilaterally, is apt.<br />

Among the studies aimed at understanding<br />

hyperfunctional behavior, most (n = 4) evaluated both the<br />

IH and SH groups, and regarded the cervical muscles as the<br />

sternocleidomastoid (SCM), scalene (SC), and trapezius<br />

(TR) muscles. Some works (n = 2) evaluated the thoracic<br />

respiratory muscles, including the rectus abdominis (RA)<br />

and major pectoralis (MP) (<strong>17</strong>,19),during singing, detecting<br />

differences in EA according to the tasks performed. Muscles<br />

of the orofacial region, such as the MA and LO, were also<br />

evaluated (8,15,25).<br />

It is interesting to mention that not all studies jointly<br />

assessed the SH and IH muscles, and investigators<br />

sometimes elected one or the other group for reasons that<br />

were not clarified in the methodology. However,<br />

investigation of the traction functions that such antagonistic<br />

muscles exert on the larynx during phonation and swallowing<br />

could extend the scope of projects studying the<br />

biomechanics of these functions. In addition, given the<br />

diversity of the tasks performed (emission of vowels,<br />

speaking, reading, and singing, with all of these tasks<br />

sometimes being requested in the same study), the reports<br />

investigated were too succinct in describing the occurrence<br />

of possible mechanical artifacts caused by the dynamics of<br />

emissions or difficulties associated with sustaining the<br />

electrodes in place during execution of these tasks.<br />

The first classical studies (10,11) using SEMG to<br />

assess phonation in people with dysphonia, as well as the<br />

findings of Silvério (7),Hocevar-Boltezar, Janko, andZargi<br />

(8), and Neli (27), showed differences in the EA of muscles<br />

between dysphonic and non-dysphonic subjects, with<br />

activity being higher in dysphonia. This indicates that<br />

dysphonic and non-dysphonic subjects actually differ in<br />

their muscular activities. However, the results of more<br />

recent studies (13,26) contraindicate the use of SEMG as a<br />

tool to assess vocal hyperfunction and indicate that it may<br />

not be used for diagnosis of muscle tension dysphonia<br />

(MTD).Even if SEMG has no sensitivity or predictive value<br />

for the diagnosis of hyperfunction, study results suggesting<br />

different responses among different groups, including<br />

dysphonic subjects, singers and laryngectomized patients<br />

(2,3,7-11,15-21,23,25,27), suggest that this procedure has<br />

the ability to evaluate behavior in an inter- and intra-subject<br />

manner, and may be used as a benchmark for clinical<br />

outcomes.<br />

Despite the methodological limitations of the studies<br />

analyzed, the technical evolution of SEMG measurement<br />

has involved some important factors that deserve<br />

consideration. For example, signal normalization is a technical<br />

recommendation that was used by 11 (40.7%) of the 27<br />

studies included in this review. During SEMG, it is<br />

recommended to use a benchmark of muscle contraction<br />

from each person evaluated; this is known as signal<br />

normalization and reduces inter- and intra-subject variability.<br />

There are several ways to normalize a SEMG signal: by<br />

using the maximum peak of electrical activity, by using the<br />

maximal voluntary contraction (MVC) or submaximal<br />

voluntary contraction (SMC), and through the mean EA.<br />

When evaluating muscles of low caliber such as the SH,<br />

IH,SC, LO, or MA muscles, the occurrence of crosstalk (i.e.,<br />

interference by signals from adjacent muscles) must be<br />

minimized to allow greater reliability in the capture and<br />

normalization of signals.<br />

The articles studied in the current review used<br />

several methods of normalization, including maximal<br />

voluntary activation (MVA) or MVC (9,10,12,13,15,18,20),<br />

SMC (2,10), peak activity (14),and activity at rest (11).<br />

MVC was the most frequently used method. This was<br />

accomplished in most studies by means of manual counterresistance<br />

or resistance against a static object (with platform<br />

to support the chin), and aimed to provide maximum<br />

activation of the SH, IH, SC, and SCM muscles. The<br />

extensive work of Stepp (28), which characterizes the<br />

clinical use of SEMG in studies on speech and swallowing,<br />

proposes the standardization of the methodological steps<br />

for use of this tool. Stepp also reports the use of normalization<br />

through MVC by various maneuvers, emphasizing manual<br />

counter-resistance (4,6,12,15,28).Despite the evidence<br />

provided by these articles, it is pertinent to note that the<br />

use of manual counter-resistance or resistance against an<br />

object may introduce a possible bias, given the possibility<br />

of recruitment of muscles adjacent to those of interest and<br />

thus the unwanted occurrence of crosstalk. This possibility<br />

gains strength when antagonistic muscle groups are being<br />

evaluated, as in the case of the SH and IH muscles.<br />

According to the studies mentioned above, the same<br />

maneuver was used to normalize the signals of different<br />

muscles. However, the use of the same maneuver to<br />

normalize all muscles being tested should be questioned.<br />

If, for example, the SCM has greater EA because it is larger<br />

and more robust, using the same normalizing maneuver for<br />

smaller muscles may result in the possibility that the SCM<br />

and perhaps the SC are also recruited for the normalization<br />

task. One measure that could minimize this potential bias<br />

would be to seek the maximum activation of specific<br />

muscle groups; since specific muscle groups have distinct<br />

and physiologically defined functions, this seems feasible.<br />

For the SCM, this would only be reliably achieved through<br />

some kind of biofeedback control that ensured the level of<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 329-339, Jul/Aug/September - 2013.<br />

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Balata et al.<br />

contraction performed was within the desired threshold<br />

(50% less than the maximum level), a precaution that was<br />

taken in 2 previous studies (2,10).<br />

Regarding normalization by the peak in speech<br />

emissions (reading, sentences, singing, and automatic<br />

sequences), this peak may be presented during the episode<br />

of respiratory pause and thus may not characterize the<br />

maximum phonation peak. In some situations, it may not<br />

even be present, as in the occurrence of an abrupt vocal<br />

attack for example.<br />

The studies investigated came to the conclusion<br />

that normality standards for the EA of the muscles involved<br />

in speech phonation could be established, but these<br />

standards were not identified in any of the studies.At first,<br />

the definition of these parameters of normality seemed to<br />

be important for consistency among studies, in order to<br />

allow safe evaluation of similar cases and even evaluation<br />

of those with different etiology. However, SEMG does not<br />

seem to allow the definition of such standards, as this type<br />

of electrophysiological evaluation of phonation is subject<br />

to individual variables such as muscle conditioning, fat,<br />

sagging dermis, and teeth arch conditions. These variables<br />

are better controlled through the use of proper<br />

normalization, whereby the subject becomes an internal<br />

reference.<br />

Although it has been advocated that the advent of<br />

high-density SEMG has contributed to the study of the<br />

action potentials of motor units, which was only possible<br />

previously using the invasive method of EMG, SEMG is not<br />

yet considered to be a tool for clinical implementation<br />

(29).Even so, it should be noted that despite the fact that<br />

methodological differences between studies hinder<br />

comparisons, the most recent studies, which are also the<br />

most numerous, use MVA normalization, which indicates a<br />

consistency in adopting this method. However, it should<br />

be assumed that normalization still constitutes a limitation<br />

of SEMG, since the maneuvers used for normalization did<br />

not differ between the muscles evaluated, which seems to<br />

create an important bias.<br />

allocation, tasks to be performed, forms of assessment,<br />

signal treatment and analysis, and especially the most<br />

appropriate method of normalization. Finally, while<br />

acknowledging that SEMG has limitations and requires<br />

technical care during its application and analysis, we<br />

believe that this is a procedure that provides quantitative<br />

information for vocal assessment and allows objective<br />

paradigms for understanding the muscles involved in this<br />

function.<br />

CONCLUSION<br />

The current state of knowledge regarding the use of<br />

SEMG for the assessment of phonation confirms the clinical<br />

applicability of this tool, as published studies demonstrate<br />

differences in EA between groups. However, SEMG appears<br />

to have no predictive value as a diagnostic test. The<br />

standardization of assessment techniques should be<br />

established in order to enable comparisons among future<br />

studies.<br />

ACKNOWLEDGEMENTS<br />

The researchers thank the National Council for<br />

Scientific and Technological Development for financial<br />

support.<br />

The current review identifies key gaps in the<br />

knowledge regarding use of SEMG for evaluation of<br />

phonation, and highlights the following challenges for<br />

future studies: normalization of electrode allocation by<br />

region and laterality; establishment of maneuvers for MVA<br />

to allow normalization by muscle (for antagonists in particular);<br />

and more robust studies with randomized control<br />

groups. However, the studies investigated point to a<br />

promising future regarding SEMG use. There is a mandatory<br />

requirement for the definition and adoption of an<br />

international system of assessment that establishes criteria<br />

for technical characteristics such as sites of electrode<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 329-339, Jul/Aug/September - 2013.<br />

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J. Clinical applications of high-density surface EMG: A<br />

systematic review. J Electromyogr Kinesiol. 2006;16:<br />

586–602.<br />

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339


Review Article<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):340-343.<br />

DOI: 10.7162/S1809-977720130003000015<br />

Middle ear adenoma with neuroendocrine differentiation: relate of two<br />

cases and literature review<br />

Aline Gomes Bittencourt 1 , Robinson Koji Tsuji 2 , Francisco Cabral Junior 3 , Larissa Vilela Pereira 3 , Anna Carolina de Oliveira Fonseca 1 ,<br />

Venâncio Alves 4 , Ricardo Ferreira Bento 5 .<br />

1) Otolaryngologist, Ph.D. Student. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

2) M.D., Ph.D. - Associated Doctor. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

3) M.D., Otolaryngology Resident. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

4) M.D. Ph.D. Professor and Chairman - Department of Patology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

5) M.D. Ph.D. Professor and Chairman - Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

Institution: Departamento de Otorrinolaringologia, Hospital das Clínicas, Universidade de São Paulo.<br />

São Paulo / SP - Brazil.<br />

Mailing address: Aline Gomes Bittencourt - Departamento de Otorrinolaringologia - Hospital das Clínicas - Universidade de São Paulo - Av. Dr. Enéas de Carvalho Aguiar,<br />

255, 6º andar/sala 6167 - São Paulo / SP – Brazil / Zip code: 05403-000 - Telephone: (+ 55 11) 3088 0299 - E-mail: alinebittencourt@hotmail.com<br />

Article received on December 20 th , 2012. Article accepted on February 18 th , 2013.<br />

SUMMARY<br />

Introduction: Adenomas with neuroendocrine differentiation are defined as neuroendocrine neoplasms, and they are rarely<br />

found in the head and neck.<br />

Objective: To describe two cases of a middle ear adenoma with neuroendocrine differentiation, with a literature review.<br />

Case Report: Patient 1 was a 41-year-old woman who presented with a 3-year history of left aural fullness associated with<br />

ipsilateral “hammer beating” tinnitus. Patient 2 was a 41-year-old male who presented with unilateral conductive hearing loss.<br />

Conclusion: Adenoma with neuroendocrine differentiation of the middle ear is a rare entity, but it should be considered in<br />

patients with tinnitus, aural fullness, and a retrotympanic mass and remembered as a diferential diagnosis of tympanic paraganglioma.<br />

Keywords: Neuroendocrine Tumors; Hearing Loss; Adenoma.<br />

INTRODUCTION<br />

Neuroendocrine cells are generally present in the<br />

respiratory epithelium as well as in the bronchial and<br />

intestinal mucosa. A few of these cells are also associated<br />

with epithelial structures found throughout the body,<br />

including the head and neck. Adenomas with<br />

neuroendocrine differentiation are defined as<br />

neuroendocrine neoplasms, and they are rarely found in<br />

the head and neck. When they occur in this region, they are<br />

more commonly found in the larynx (1). They usually grow<br />

in the lungs or gastrointestinal tract (1, 2).<br />

CASE REPORTS<br />

Patient 1 was a 41-year-old woman who presented<br />

with a 3-year history of left aural fullness associated with<br />

ipsilateral “hammer beating” tinnitus. Over the following 8<br />

months, the patient developed left-side otalgia and was<br />

treated with systemic antibiotics but without any<br />

improvement.<br />

The patient consulted a colleague who raised the<br />

possibility of a tympanic paraganglioma. Computed<br />

tomography (CT) of the temporal bones revealed a material<br />

with a soft tissue density that diffusely obliterated the<br />

left mastoid cells, but with no signs of ossicular chain<br />

erosion (Figure 1). Angiography of the temporal bone<br />

showed a lobulated solid lesion with defined limits and<br />

contrast uptake within the left middle ear and the<br />

epitympanic recess.<br />

The patient consulted our institution when her<br />

symptoms persisted. She had no complaints of hearing loss<br />

or otorrhea. On examination, the patient was found to be<br />

in good general condition and without neurological deficits.<br />

Otoscopy of the right ear was normal, but a retrotympanic,<br />

non-pulsatile mass was detected in the left ear (Figure 2).<br />

Figure 3 demonstrates her puretone audiometry, with a<br />

10dB-gap in the left ear. She subsequently underwent a left<br />

radical mastoidectomy with total resection of the lesion.<br />

Intraoperatively, the tumor had a fibrous aspect and filled<br />

the antrum and the entire tympanic cavity, involving the<br />

ossicular chain. The histopathological study was compatible<br />

with middle ear adenoma with neuroendocrine<br />

differentiation.<br />

Patient 2 was a 41-year-old man who presented<br />

with unilateral conductive hearing loss. CT scan showed a<br />

middle ear mass in the promontory mimicking a tympanic<br />

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Middle ear adenoma with neuroendocrine differentiation: relate of two cases and literature review.<br />

Bittencourt et al.<br />

A B C<br />

Figure 1. Computed tomography of the temporal bones revealed a material with a soft tissue density that diffusely obliterated<br />

the left mastoid cells, but with no signs of ossicular chain erosion. A. Coronal B. Axial C. Transverse<br />

Figure 2. Otoscopy of the left ear demonstrating a retrotympanic<br />

non-pulsatile mass.<br />

Figure 3. Pure tone audiometry showing a gap in the left ear.<br />

A<br />

B<br />

Figure 4. Computed tomography of the temporal bones revealing a small soft tissue<br />

density material in the left tympanic cavity. A. Coronal B. Axial<br />

paraganglioma (Figure 4). Magnetic ressonance imaging<br />

(MRI) of the temporal bones revealed a small mass in the<br />

left tympanic cavity with isosignal in T1 and post-contrast<br />

enhancement on T2 sequences(Figure 5). The patient<br />

underwent surgical intervention through a supra-meatal<br />

approach that completely removed the mass. The tumor<br />

was brownish and elastic, involved the ossicles and extended<br />

to the promontory. Histology and immunohistochemical<br />

reactions were also compatible with middle ear adenoma<br />

with neuroendocrine differentiation (Figure 6).<br />

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Middle ear adenoma with neuroendocrine differentiation: relate of two cases and literature review.<br />

Bittencourt et al.<br />

A<br />

B<br />

Figure 5. A: MRI of the temporal bones revealing a small mass in the left tympanic<br />

cavity with isosignal in T1 (A) and post-contrast enhancement on T2.<br />

.<br />

A<br />

B<br />

C<br />

D<br />

Figure 6. A: Well-differentiated papillary/trabecular/solid epithelial neoplasm composed of medium-sized cells. Central<br />

nuclei presented salt-and-pepper – like chromatin, and granular eosinophilic cytoplasm. Some neoplastic cells were seen<br />

amidst a fibrotic stroma. HE stain, original magnification x200. B-C: Immunohistochemical identification keratins marked<br />

by the antibodies AE1+AE3 and most of them were reactive for neuroendocrine markers chromogranin and<br />

synaptophysin. Original magnification x100. D: Ki-67 Ag was expressed in very low level (< 1 % of the cells). Original<br />

magnification x400.<br />

REVIEW OF THE LITERATURE<br />

In 1980, Murphy et al. first reported a middle ear<br />

adenoma with neuroendocrine differentiation, also called<br />

carcinoid tumor, as various denominations are used for<br />

these neoplasias, reflecting the controversy between its<br />

presumed histogenesis and differentiation (3). The lesion<br />

had adenomatous histological characteristics, but with<br />

ultrastructural neuroendocrine differentiation (1, 4). Since<br />

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Middle ear adenoma with neuroendocrine differentiation: relate of two cases and literature review.<br />

Bittencourt et al.<br />

then, approximately 40 cases have been described in the<br />

English literature (1).<br />

The incidence of this tumor in the middle ear is<br />

difficult to establish since it may be confused with<br />

adenomatous tumors, ceruminomas, and even with other<br />

tumors, such as paragangliomas (1).<br />

Clinically, it presents with conductive hearing loss<br />

and ear fullness. Less frequently, patients may complain of<br />

tinnitus, headache, and even facial paralysis (5). In most<br />

cases, otoscopy demonstrates an intact tympanic membrane<br />

with a retrotympanic mass, as occurred in this case.<br />

Carcinoid syndrome (flushing, sweating, and diarrhea) is<br />

not found in these patients because of the small production<br />

of vasoactive peptides. Distant metastases are rare events<br />

(1, 5). Imaging studies consistently show a locally infiltrative<br />

mass and may show invasion of the ossicles (4).<br />

Middle ear adenoma with neuroendocrine<br />

differentiation can be distinguished from adenomatous<br />

tumors (adenomas, adenocarcinomas, and adenoid cystic<br />

carcinomas), ceruminomas, and paragangliomas by their<br />

light microscopic appearance, argyrophilic granules, and<br />

ultrastructural evidence of neurosecretory granules, as well<br />

as by immunohistochemical detection of various<br />

neuroendocrine markers. These include neuron-specific<br />

enolase, chromogranin, synaptophysin, serotonin, glucagon,<br />

insulin, gastrin, vasoactive intestinal polypeptide, pancreatic<br />

polypeptide, and calcitonin (4).<br />

DISCUSSION<br />

We reported two cases of middle ear adenoma with<br />

neuroendocrine differentiation wich presented with<br />

unspecific clinical manifestations. In both cases, the<br />

radiological features were very similar to tympanic<br />

paraganglioma, wich is the most frequent tumoral lesion of<br />

the middle ear. The final diagnosis was provided by<br />

histological and immunohistochemical analysis of the tumor.<br />

The recommended treatment is complete surgical<br />

excision of the lesion and the technique used is determined<br />

by the size and involvement of adjacent structures (1).<br />

Radiotherapy is not indicated since the tumor has poor<br />

radiosensitivity (1). The prognosis is good, however, a<br />

rigorous postoperative follow-up is indicated for all patients<br />

(3).<br />

Our patients underwent surgical treatment and<br />

clinical follow-up in conjunction with the Oncology<br />

department. One of the patients (patient 1) had imaging<br />

studies performed 18 months after surgery, wich confirmed<br />

that there were no signs of disease recurrence.<br />

FINAL COMMENTS<br />

Middle ear adenoma with neuroendocrine<br />

differentiation is a rare entity, but it should be considered<br />

as a possible diagnosis in patients with hearing loss, tinnitus,<br />

aural fullness, and a retrotympanic mass.<br />

REFERENCES<br />

1. Ferlito A, Devaney K, Rinaldo A. Primary carcinoid tumor<br />

of the middle ear: A potentially metastasizing tumor. Acta<br />

Otolaryngol. 2006;126(3):228-31.<br />

2. Pellini R, Ruggieri M, Pichi B, et. al. A case of cervical<br />

metastases from temporal bone carcinoid. Head Neck.<br />

2005;27(7):644-47.<br />

3. Becker CG, Barra IM, Pimenta HHO, Crosara PFTB, Ribeiro<br />

CA, Castro LPF. Adenoma da orelha média: relato de caso.<br />

Rev Bras Otorrinolaringol. 2004;70(4):551-4.<br />

4. Knerer B, Matula C, Youssefzadeh S, et al. Treatment of<br />

a local recurrence of a carcinoid tumor of the middle ear by<br />

extended subtotal petrosectomy. Eur Arch Otorhinolaryngol.<br />

1998;255(2):57-61.<br />

5. Shibosawa E, Tsutsumi K, Ihara Y, et al. A case of carcinoid<br />

tumor of the middle ear. Ausis, Nasus, Larynx. 2003;30<br />

Suppl:S99-S102.<br />

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343


Case Report<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):344-346.<br />

DOI: 10.7162/S1809-977720130003000016<br />

Bullous Systemic Lupus Erythematosus: Case report<br />

Ivan Dieb Miziara 1 , Ali Mahmoud 2 , Azis Arruda Chagury 3 , Ricardo Dourado Alves 4 .<br />

1) Associate Professor in the Department of Otorhinolaryngology, School of Medicine, University of São Paulo, São Paulo, Brazil.<br />

2) Otolaryngologist in the Department of Otorhinolaryngology, School of Medicine, University of São Paulo, São Paulo, Brazil.<br />

3) Specialist in Otolaryngology, School of Medicine, University of São Paulo, São Paulo, Brazil.<br />

4) Resident in Otolaryngology, School of Medicine, University of São Paulo, São Paulo, Brazil.<br />

Institution: Study from the Division of Clinical Otorhinolaryngology, Clinic of Stomatology, Hospital of the Faculty of Medicine, University of São Paulo.<br />

São Paulo / SP - Brazil.<br />

Mailing address: ENT Hospital - Azis Arruda Chagury - Avenue Dr. Enéas de Carvalho Aguiar, 155 - São Paulo / SP - Brazil - Zip code: 05403-000 - Telephone:<br />

(+55 11) 3069-6226 - E-mail: azischagury@gmail.com<br />

Article received on September <strong>17</strong> th , 2011. Article accepted on February 5 th , 2012.<br />

SUMMARY<br />

Introduction: Bullous systemic lupus erythematosus (BSLE) is an autoantibody-mediated disease with subepidermal blisters.<br />

It is a rare form of presentation of SLE that occurs in less than 5% of cases of lupus.<br />

Case Report: A 27-year-old, female, FRS patient reported the appearance of painful bullous lesions in the left nasal wing<br />

and left buccal mucosa that displayed sudden and rapid growth. She sought advice from emergency dermatology staff 15<br />

days after onset and was hospitalized with suspected bullous disease. Intravenous antibiotics and steroids were administered<br />

initially, but the patient showed no improvement during hospitalization. She displayed further extensive injuries to the trunk,<br />

axillae, and vulva as well as disruption of the bullous lesions, which remained as hyperemic scars. Incisional biopsy of a<br />

lesion in the left buccal mucosa was performed, and pathological results indicated mucositis with extensive erosion and the<br />

presence of a predominantly neutrophilic infiltrate with degeneration of basal cells and apoptotic keratinocytes. Under direct<br />

immunofluorescence, the skin showed anti-IgA, anti-IgM, and anti-IgG linear fluorescence on the continuous dermal side<br />

of the cleavage. Indirect immunofluorescence of the skin showed conjugated anti-IgA, was anti-IgM negative, and displayed<br />

pemphigus in conjunction with anti-IgG fluorescence in the nucleus of keratinocytes, consistent with a diagnosis of bullous<br />

lupus erythematosus.<br />

Discussion: BSLE is an acquired autoimmune bullous disease caused by autoantibodies against type VII collagen or other<br />

components of the junctional zone, epidermis, and dermis. It must be differentiated from the secondary bubbles and vacuolar<br />

degeneration of the basement membrane that may occur in acute and subacute cutaneous lupus erythematosus.<br />

Keywords: Lupus Erythematosus, Systemic; Stomatitis; Mucositis<br />

INTRODUCTION<br />

Bullous systemic lupus erythematosus (BSLE) is an<br />

autoantibody-mediated disease with subepidermal blisters.<br />

It is a rare presentation of SLE occurring in less than 5% of<br />

lupus cases (1). A study conducted in 3 regions of France<br />

reported an incidence of 0.2 cases per million inhabitants<br />

(2). Although the disease affects both men and women<br />

regardless of race or age, it occurs more frequently in<br />

black women between the second and third decades of<br />

life.<br />

Clinically, BSLE presents as tense bullous lesions<br />

that can be serous or hemorrhagic and spread rapidly to<br />

all parts of the body, although lesions are mainly found on<br />

the trunk and in sun-exposed areas; they can also affect<br />

mucous membranes. These lesions often indicate lupus<br />

manifestation (1). However, they must be differentiated<br />

from other bullous dermatoses such as epidermolysis<br />

bullosa acquisita (EBA), dermatitis herpetiformis, bullous<br />

pemphigoid, and linear IgA bullous dermatosis (3).<br />

In this paper, we report the case of a young female<br />

patient who suddenly developed bullous disease and was<br />

admitted to hospital because of severe oral lesions. After<br />

immunofluorescent examination, a diagnosis of bullous<br />

lupus erythematosus was made.<br />

CASE REPORT<br />

Here, we report a 27-year-old, single, catholic,<br />

female, born in Sierra Taboão and raised in Embu. The<br />

patient reported emergence of painful bullous lesions on<br />

the nasal ala and left buccal mucosa that displayed sudden<br />

and rapid growth (Fig. 1). She sought advice from the<br />

emergency dermatology staff 15 days after the onset of<br />

symptoms and was hospitalized with suspected bullous<br />

disease. General examinations were normal and her<br />

leukocyte levels were unchanged. Upon hospitalization,<br />

intravenous steroids were introduced but the patient showed<br />

no clinical improvement. She displayed further extensive<br />

injuries to the trunk, axillae, and vulva as well as rupture of<br />

the bullous lesions, which remained as reddened scars.<br />

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Bullous Systemic Lupus Erythematosus: Case report.<br />

Miziara et al.<br />

Figure 1. Bullous lesion in the right lower lip.<br />

Figure 2. Predominantly neutrophilic inflammatory infiltrate<br />

with hydropic degeneration of the basal layer and apoptotic<br />

keratinocytes.<br />

Figure 4. Lack of oral lesions, without mucosal scarring.<br />

Figure 3. Direct immunofluorescence of the skin showing<br />

linear fluorescence on the continuous dermal side of the<br />

cleavage.<br />

We performed an incisional biopsy of a lesion in the<br />

left buccal mucosa. The pathology results indicated mucositis<br />

with extensive erosion and the presence of a predominantly<br />

neutrophilic inflammatory infiltrate with degeneration and<br />

apoptosis of basal layer keratinocytes (Fig. 2). Direct skin<br />

immunofluorescence showed anti-IgA, anti-IgM, and anti-<br />

IgG linear fluorescence on the continuous dermal side of<br />

the cleavage (Fig. 3).<br />

Indirect immunofluorescence of the skin showed<br />

conjugated anti-IgA, was anti-IgM negative, and indicated<br />

pemphigus. It also displayed anti-IgG conjugated with<br />

fluorescence in the nucleus of keratinocytes, confirming<br />

the diagnosis of bullous lupus erythematosus.<br />

As initial treatment, we administered 100 mg<br />

dapsone, 250 mg chloroquine, 50 mg azathioprine, and<br />

50 mg prednisone. Although this resulted in improvement<br />

of the lesions, there was persistence of some scarring<br />

plaques on the trunk and lower lip. The patient was<br />

followed up for 6 months. During this period, she remained<br />

clinically stable, without active lesions in the oral mucosa<br />

and lips (Fig. 4), and we gradually reduced her dose of<br />

prednisone.<br />

DISCUSSION<br />

In 1973, Pedro and Dahl (4) described the first case<br />

of BSLE. After this, several cases with similar characteristics<br />

were reported. BSLE patients produce autoantibodies that<br />

recognize type VII collagen, a major component of anchoring<br />

fibrils, which play an important role in dermoepidermal<br />

adhesion.<br />

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Bullous Systemic Lupus Erythematosus: Case report.<br />

Miziara et al.<br />

Chan and colleagues (5) identified further<br />

autoantibodies reacting to multiple basement membrane<br />

components including bullous pemphigoid antigen 1,<br />

laminin-5, and laminin-6 in patients with BSLE. This<br />

hyperimmune state seems to be associated with the gene<br />

for major histocompatibility complex HLA-DR2 (4).<br />

The clinical manifestations of BSLE are characterized<br />

by vesicles, serous blisters, or hemorrhagic content in the<br />

face, neck, and trunk. Lesions can be located in areas both<br />

exposed and unexposed to the sun. They may be<br />

accompanied by mild to severe itching and can affect<br />

mucous membranes. BSLE lesions may heal completely, or<br />

result in hypo-or hyperpigmentation or scarring (6).<br />

The clinical picture of BSLE is similar to bullous<br />

pemphigus, being distinguished by indirect<br />

immunofluorescence. Histologically, the condition presents<br />

as subepidermal blisters accompanied by neutrophil<br />

microabscesses in the papillary dermis and dermal edema.<br />

There are also large deposits of mucin and the absence of<br />

eosinophils, which helps to differentiate between dermatitis<br />

herpetiformis Duhring, linear IgA dermatosis, and EBA (6).<br />

In BSLE, direct immunofluorescence shows deposits<br />

of immune complexes (IgG, IgA, IgM, and complement)<br />

along the basement membrane that can be granular to<br />

linear (7).<br />

Using indirect immunofluorescence, and according<br />

to the autoantibodies present, BSLE can be classified into<br />

3 subtypes: type 1, which reacts against collagen VII; type<br />

2, where the location of the antigen is undefined or the<br />

dermal antigen is one other than type VII collagen; and<br />

type 3, where the antigen is epidermal (6).<br />

BSLE responds well to treatment with dapsone,<br />

which differs from EBA. In some cases where high doses of<br />

corticosteroids alone were used for control of visceral<br />

manifestations, there was no improvement of skin lesions<br />

until the introduction of dapsone. It is expected that within<br />

24–48 h of the introduction of dapsone, the emergence of<br />

new bubbles will be stopped, and total regression should<br />

occur within weeks. In cases that do not respond to<br />

dapsone, the use of prednisone and azathioprine is<br />

suggested. BSLE may regress completely and independently<br />

of systemic involvement without recurrence (8).<br />

REFERENCE<br />

1. M Tincopa, Puttgen KB, Sule S, Cohen BA, Gerstenblith<br />

MR. Bullous lupus: an unusual initial presentation of systemic<br />

lupus erythematosus in an adolescent girl. Pediatr Dermatol.<br />

2010 Jul-Aug;27(4):373-6.<br />

2. Bernard P, Vaillant L, Labeille B, et al. Incidence and<br />

distribution of subepidermal autoimmune bullous skin<br />

diseases in three French regions. Bullous Diseases French<br />

Study Group. Arch Dermatol. 1995;131(1):48-52.<br />

3. Vieira FMJ, Oliveira ZNP. Bullous systemic lupus<br />

erythematosus. An Bras Dermatol. 1998;73:143-7.<br />

4. Peter SD, Dahl MV. Direct immunofluorescence of Bullous<br />

systemic lupus erythematosus. Arch Dermatol.<br />

1973;107:118-20.<br />

5. Chan LS, LaPiere JC, Chen M, Traczyk T, Mancini AJ, Paller<br />

AS, et al. Bullous systemic lupus erythematosus with<br />

autoantibodies recognizing multiple skin basement<br />

membrane components, bullous pemphigoid antigen 1,<br />

laminin-5, laminin-6, and type VII collagen. Arch Dermatol.<br />

1999;135:569-73.<br />

6. Obermoser G, Sontheimer RD, Zelger B. Overview of<br />

common, rare and atypical manifestations of cutaneous lupus<br />

erythematosus and histopathological correlates. Lupus. 2010<br />

Aug;19(9):1050-70.<br />

7. Cato, Ellen Erie et al. Bullous systemic lupus erythematosus<br />

associated with lupus nephritis: report of two cases. An Bras<br />

Dermatol. 2007;82(1):57-61.<br />

8. Vassileva S. Bullous systemic lupus erythematosus. Clin<br />

Dermatol. 2004 Mar-Apr;22(2):129-38.<br />

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Case Report<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):347-350.<br />

DOI: 10.7162/S1809-9777201300030000<strong>17</strong><br />

Eagle’s Syndrome<br />

Thaís Gonçalves Pinheiro 1 , Vítor Yamashiro Rocha Soares 2 , Denise Bastos Lage Ferreira 3 , Igor Teixeira Raymundo 3 ,<br />

Luiz Augusto Nascimento 4 , Carlos Augusto Costa Pires de Oliveira 5 .<br />

1) MD. Fellowship in Laryngology, Department of Otorhinolaryngology, University of Sao Paulo.<br />

2) MD. Ph.D. Student in Health Sciences, Department of Otorhinolaryngology - Head and Neck Surgery, University of Brasilia.<br />

3) MD. Attending Physician, Department of Otorhinolaryngology - Head and Neck Surgery, University of Brasilia.<br />

4) MD, PhD. Adjunct Professor, Department of Otorhinolaryngology - Head and Neck Surgery, University of Brasilia.<br />

5) MD, PhD. Full Professor, Department of Otorhinolaryngology - Head and Neck Surgery, University of Brasilia.<br />

Institution: University of Brasilia – University of Brasilia Hospital, Department of Otorhinolaryngology - Head and Neck Surgery.<br />

SGAN, Via L2 Norte, Quadra 604/605, Asa Norte - Ambulatório II, Brasilia/DF – Brazil, ZC: 70.840-050.<br />

Mailing address: Vítor Yamashiro Rocha Soares - SGAN, Via L2 Norte, Quadra 604/605 - HUB - Anexo III, Apart. 20 - Asa Norte - Brasília/DF - Brazil, Zip code:<br />

70840-050 - E-mail: vyrsoares@gmail.com<br />

Article received on October 5 th , 2011. Article accepted on November 16 th , 2011.<br />

SUMMARY<br />

Introduction: Eagle’s syndrome is characterized by cervicopharyngeal signs and symptoms associated with elongation of the<br />

styloid apophysis. This elongation may occur through ossification of the stylohyoid ligament, or through growth of the apophysis<br />

due to osteogenesis triggered by a factor such as trauma. Elongation of the styloid apophysis may give rise to intense facial<br />

pain, headache, dysphagia, otalgia, buzzing sensations, and trismus. Precise diagnosis of the syndrome is difficult, and it is<br />

generally confounded by other manifestations of cervicopharyngeal pain.<br />

Objective: To describe a case of Eagle’s syndrome.<br />

Case Report: A 53-year-old man reported lateral pain in his neck that had been present for 30 years. Computed tomography<br />

(CT) of the neck showed elongation and ossification of the styloid processes of the temporal bone, which was compatible with<br />

Eagle’s syndrome. Surgery was performed for bilateral resection of the stylohyoid ligament by using a transoral and endoscopic<br />

access route. The patient continued to present pain laterally in the neck, predominantly on his left side. CT was performed again,<br />

which showed elongation of the styloid processes. The patient then underwent lateral cervicotomy with resection of the stylohyoid<br />

process, which partially resolved his painful condition.<br />

Final Comments: Patients with Eagle’s syndrome generally have a history of chronic pain. Appropriate knowledge of this<br />

disease is necessary for adequate treatment to be provided. The importance of diagnosing this uncommon and often unsuspected<br />

disease should be emphasized, given that correct clinical-surgical treatment is frequently delayed. The diagnosis of Eagle’s<br />

syndrome is clinical and radiographic, and the definitive treatment in cases of difficult-to-control pain is surgical.<br />

Keywords: Neck Pain; Osteogenesis; Oral Surgical Procedures.<br />

INTRODUCTION<br />

Eagle’s syndrome is characterized by<br />

cervicopharyngeal signs and symptoms associated with<br />

elongation of the styloid apophysis (1). The styloid<br />

apophysis consists of a bone projection originating in the<br />

tympanic portion of the temporal bone, which is<br />

approximately 25 mm in length (2). Elongation of the<br />

styloid apophysis may occur through ossification of the<br />

stylohyoid ligament, or through growth of the apophysis<br />

due to osteogenesis triggered by a factor such as trauma<br />

(1).<br />

Elongation of the styloid apophysis or calcification<br />

of the stylohyoid ligament may give rise to intense facial<br />

pain, headache, dysphagia or odynophagia, otalgia, buzzing<br />

sensations, and trismus (1-4). Precise diagnosis of the<br />

syndrome is difficult. It is generally confounded by<br />

temporomandibular joint dysfunction, chronic tonsillitis,<br />

migraine, cluster headache, glossopharyngeal and trigeminal<br />

neuralgia, myofascial pain dysfunction syndrome, pain<br />

secondary to unerupted or impacted third molars, neck<br />

arthritis, and tumors of the base of the tongue. Finger<br />

palpation of the palatine tonsil increases the pain associated<br />

with Eagle’s syndrome and corroborates its diagnosis. In<br />

addition to a clinical history and physical examination,<br />

radiological evaluation is also important for diagnosis (4).<br />

The aims of this study were to describe a case of<br />

Eagle’s syndrome followed up at the Otorhinolaryngology<br />

- Head and Neck Surgery Service and to conduct a critical<br />

review of the literature.<br />

CASE REPORT<br />

The patient was a 53-year-old man who reported<br />

having had dysphagia as well as a stabbing pain laterally<br />

in his neck and bilateral cervical pain in his<br />

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temporomandibular joint for 30 years. The pain extended<br />

to the deltoid region and, although intermittent, occurred<br />

every day along with hardening of the left side of the neck<br />

and episodic frontal headache. The condition had<br />

progressively worsened over the preceding 10 years, in<br />

terms of both intensity and frequency. The patient<br />

reported that the pain worsened with heat, especially<br />

with exposure to sunlight, and when lying down on his<br />

back. Clinical treatment using analgesics, anti-inflammatory<br />

agents, or muscle relaxants did not alleviate the pain. The<br />

patient said that he had not had any previous pathological<br />

conditions, except for an appendectomy 32 years earlier.<br />

There were no palpable enlarged lymph nodes in the<br />

neck region. Oral examination showed a hardened lesion<br />

in the posterior tonsillar pillar measuring 15 mm and a<br />

retromolar bone spur on the left side.<br />

Radiography and computed tomography of the<br />

neck showed elongation and ossification of the styloid<br />

processes of the temporal bone. The right styloid process<br />

measured 5.7 cm and the left measured 4.9 cm, which<br />

was compatible with Eagle’s syndrome (Figures 1A and<br />

1B).<br />

Surgery was performed for bilateral resection of the<br />

stylohyoid ligament by using a transoral endoscopic access<br />

route. However, the patient continued to present<br />

preauricular pain with a burning sensation irradiating to the<br />

sides of the neck and temporal region, predominantly on<br />

his left side. Another computed tomography scan was<br />

produced, which showed elongation and ossification of the<br />

styloid processes of the temporal bone, with the right side<br />

measuring 4.4 cm and the left side measuring 3.9 cm. This<br />

suggested that the base of the styloid process continued to<br />

be present bilaterally. The patient then underwent lateral<br />

cervicotomy with resection of the stylohyoid process,<br />

which partially resolved his painful condition (Figures 1C<br />

and 1D).<br />

FIGURE 1. (A) Computed tomography (CT) scan of the neck showing elongation and ossification of the styloid<br />

processes of the temporal bone. The right styloid process measures 5.7 cm and the left measures 4.9 cm, which<br />

is compatible with Eagle’s syndrome. (B) CT scan of the neck after intraoral styloidectomy, showing ossification<br />

of the styloid processes. The right styloid process measures 4.4 cm and the left measures 3.9 cm, thus suggesting<br />

that the base of the styloid process is present on both sides. (C) Surgical specimen after extraoral styloidectomy:<br />

right and left styloid processes. (D) CT scan of the neck after extraoral styloidectomy, showing the absence of<br />

elongation of the styloid processes on both sides.<br />

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DISCUSSION<br />

Eagle’s syndrome is directly related to calcification<br />

of the stylohyoid ligament or elongation of the styloid<br />

process. These processes trigger symptoms such as recurrent<br />

facial pain, foreign body sensation, dysphagia, otalgia, and<br />

trismus (4). The styloid process, stylohyoid ligament, and<br />

lesser cornu of the hyoid bone form the stylohyoid complex,<br />

which originates embryologically in Reichert’s cartilage of<br />

the second branchial arch (1,2). Reichert’s cartilage has 4<br />

portions: the upper portion, from which the styloid process<br />

develops; the central portion, from which the stylohyoid<br />

ligament forms; the lower portion, from which the lesser<br />

cornu of the hyoid bone originates; and the basal portion,<br />

which gives rise to part of the hyoid bone (1,4).<br />

The reported sizes of the normal styloid process in<br />

the literature show a high degree of variability. Moffat et al<br />

conducted studies on cadavers and showed that the normal<br />

range of lengths of the styloid process was from 1.52 cm<br />

to 4.77 cm (5). In most radiographic studies, styloid<br />

processes are considered normal when they are shorter<br />

than 2.5 cm and elongated when they are longer than 4.0<br />

cm. In the latter situation, Eagle’s syndrome is very likely<br />

to be present (1). It seems that there is little correlation<br />

between the extent of ossification and the severity of<br />

symptoms (6).<br />

Several theories have been proposed to explain the<br />

elongation of the styloid process in Eagle’s syndrome,<br />

including congenital elongation resulting from persistence<br />

of a mesenchymal embryonic leaf, with the capacity to<br />

produce bone tissue in adults; reactional ossifying<br />

hyperplasia due to osteitis, periostitis, or tendinitis, caused<br />

by surgical trauma or chronic local irritation; and ossification<br />

of the stylohyoid ligament related to endocrine disorders in<br />

women at menopause, accompanied by ossification of<br />

other ligaments. Some authors have affirmed that the<br />

syndrome frequently correlates with tonsillectomy (1).<br />

However, Prasad et al did not observe this relationship in<br />

a series of 58 cases (2). The patient in the present case did<br />

not have any previous pathological history of tonsillectomy.<br />

The prevalence of elongation of the styloid process<br />

is variable. Some reports have shown that this abnormality<br />

occurs in 4% of the population, but that only 4% of such<br />

individuals present symptoms (2,4). In 1970, Kaufman et<br />

al confirmed this low rate of correlation, reporting that the<br />

styloid process was elongated in 7.3% of their patients (7).<br />

In 1979, Correl et al reported that calcification of the styloid<br />

complex or elongation of the styloid process was present<br />

in 18% of the <strong>17</strong>00 panoramic radiographs that they<br />

examined, and observed low concordance with the presence<br />

of symptoms (8). Thus, the majority of patients with an<br />

elongated styloid process are asymptomatic (4). Greater<br />

prevalence of the disease has been found in women (3:1)<br />

(2,4).<br />

The main symptoms of the syndrome include<br />

nonspecific neck pain, foreign body sensation in the<br />

pharynx, odynophagia, otalgia, pain in the<br />

temporomandibular joint, and trismus (2). Painful palpation<br />

of the tonsillar fossa, together with suggestive clinical and<br />

radiological signs, corroborates the diagnosis of Eagle’s<br />

syndrome. In addition, pain relief achieved through<br />

infiltration of local anesthetics into the tonsillar fossa is<br />

suggestive of the disease (3,6).<br />

Radiological examination can confirm the syndrome.<br />

Radiography can show the increased length of the styloid<br />

process. Currently, computed tomography is considered to<br />

be the best examination for defining the length and<br />

angulation of the styloid process, and also enables an<br />

evaluation of the anatomical relationships between the<br />

stylohyoid complex and adjacent structures (1,3,4).<br />

According to some authors, panoramic or lateral radiographs<br />

are sufficient for diagnosing the disease (2).<br />

The other conditions identified in differential<br />

diagnosis for the syndrome include temporomandibular<br />

joint dysfunction, pain secondary to unerupted or impacted<br />

third molars, glossopharyngeal and trigeminal neuralgia,<br />

migraine, neck arthritis, cluster headache, myofascial pain<br />

dysfunction syndrome, and tumors (4). Neuralgia of the IX th<br />

and V th cranial pairs consists of sudden sharp pain of short<br />

duration that arises through stimulation of the trigger zone,<br />

which is unlike that observed in Eagle’s syndrome, where<br />

the pain is dull and persistent. Disorders of the<br />

temporomandibular joint and third molar pain are confirmed<br />

through radiological examinations (1).<br />

Treatment for Eagle’s syndrome can be surgical, but<br />

is not always. Pain control may be achieved in some<br />

patients by means of oral analgesics. Some authors have<br />

suggested that transpharyngeal infiltration of steroids and<br />

local anesthetics in the tonsillar fossa should be used (1,2).<br />

The treatment of choice is styloidectomy, which can be<br />

performed transorally or extraorally. The transoral route<br />

consists of an incision in the tonsillar fossa, after tonsillectomy<br />

if necessary, which is followed by identification, separation,<br />

and excision of the styloid process. In the external technique,<br />

an incision is made in the neck, in the region proximal to<br />

the sternocleidomastoid muscle, into the hyoid bone,<br />

followed by identification, separation, and removal of the<br />

styloid process. This technique makes it possible to view<br />

the structures better, thus enabling greater resection of the<br />

styloid process (1) with less chance of deep infection in the<br />

neck (2). However, the extraoral technique requires a<br />

longer surgical recovery and it involves greater morbidity<br />

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(relating to drains, sutures, risk of facial nerve lesions, and<br />

scarring of the neck).<br />

FINAL COMMENTS<br />

Patients with Eagle’s syndrome generally have a<br />

history of chronic pain treated by several physicians.<br />

Appropriate knowledge of this disease is necessary for<br />

adequate treatment to be provided. The importance of<br />

diagnosing this uncommon and often unsuspected disease<br />

should be emphasized, given that correct clinical-surgical<br />

treatment is frequently delayed. The diagnosis of Eagle’s<br />

syndrome is clinical and radiographic, and the definitive<br />

treatment in cases of difficult-to-control pain is surgical.<br />

REFERENCES<br />

1. Fini G, Gasparini G, Filippini F, Becelli R, Marcotullio D.<br />

The long styloid process syndrome or Eagle’s syndrome. J<br />

Craniomaxillofac Surg. 2000;28:123-7.<br />

2. Prasad KC, Kamath MP, Reddy JM, Raju K, Agarwal S.<br />

Elongated styloid process (Eagle’s syndrome): a clinical<br />

study. J Oral Maxillofac Surg. 2002;60(2):<strong>17</strong>1-5.<br />

3. Beder E, Ozgursoy OB, Ozgursoy SK, Anadolu Y. Threedimensional<br />

computed tomography and surgical treatment<br />

for Eagle’s syndrome. Ear Nose Throat J. 2006;85(7):443-<br />

5.<br />

4. Mendelsohn AH, Berke GS, Chhetri DK. Heterogeneity<br />

in the clinical presentation of Eagle’s syndrome. Otolaryngol<br />

Head Neck Surg. 2006;134:389-93.<br />

5. Moffat DA, Ramsden RT, Shaw HJ. The styloid process<br />

syndrome: aetiological factors and surgical management. J<br />

Laryngol Otol. 1977;91(4):279-94.<br />

6. Diamond LH, Cottrell DA, Hunter MJ, Papage<strong>org</strong>e M.<br />

Eagle’s syndrome: a report of 4 patients treated using a<br />

modified extraoral approach. J Oral Maxillofac Surg.<br />

2001;59:1420-6.<br />

7. Kaufman SM, Elzay RP, Irish EF. Styloid process variation.<br />

Radiologic and clinical study. Arch Otolaryngol.<br />

1970;91(5):460-3.<br />

8. Correl R, Jensen J, Taylor J, et al. Mineralization of the<br />

stylohyoid-stylomandibular ligament complex: A<br />

radiographic incidence study. Oral Surg Oral Med Oral Pathol.<br />

1979;48:286-91.<br />

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Case Report<br />

Int. Arch. Otorhinolaryngol. 2013;<strong>17</strong>(3):351-355.<br />

DOI: 10.7162/S1809-977720130003000018<br />

Retrolabyrinthine approach for cochlear nerve preservation in<br />

neurofibromatosis type 2 and simultaneous cochlear implantation<br />

Ricardo Ferreira Bento 1 , Tatiana Alves Monteiro 2 , Aline Gomes Bittencourt 3 , Maria Valeria Schmidt Goffi-Gomez 4 ,<br />

Rubens de Brito 5 .<br />

1) Otolaryngologist, PhD. Professor and Chairman. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

2) Otolaryngologist. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

3) Otolaryngologist, PhD student. Neurotology Fellow. Division of Otorhinolaryngology, University of São Paulo Medical School, São Paulo, Brazil.<br />

4) Audiologist, PhD in Human Communication Disorders (Speech Pathology). Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo,<br />

Brazil.<br />

5) Otolaryngologist, PhD. Associate Professor. Department of Otolaryngology, University of São Paulo School of Medicine, São Paulo, Brazil.<br />

Institution: Division of Otorhinolaryngology, University of São Paulo Medical School.<br />

São Paulo / SP – Brazil.<br />

Mailing address: Dra. Aline Gomes Bittencourt - Departamento de Otorrinolaringologia - Hospital das Clínicas - Universidade de São Paulo - Av. Dr. Enéas de Carvalho<br />

Aguiar, 255, 6º andar/sala 6167 - São Paulo / SP – Brazil – Zip code: 05403-000 Telephone: (+ 55 11) 3088-0299 - E-mail: alinebittencourt@hotmail.com<br />

Article received on March 18 th , 2013. Article accepted on April 18 th , 2013.<br />

SUMMARY<br />

Introduction: Few cases of cochlear implantation (CI) in neurofibromatosis type 2 (NF2) patients had been reported in the<br />

literature. The approaches described were translabyrinthine, retrosigmoid or middle cranial fossa.<br />

Objectives: To describe a case of a NF2- deafened-patient who underwent to vestibular schwannoma resection via RLA with<br />

cochlear nerve preservation and CI through the round window, at the same surgical time.<br />

Resumed Report: A 36-year-old woman with severe bilateral hearing loss due to NF2 was submitted to vestibular schwannoma<br />

resection and simultaneous CI. Functional assessment of cochlear nerve was performed by electrical promontory stimulation.<br />

Complete tumor removal was accomplishment via RLA with anatomic and functional cochlear and facial nerve preservation.<br />

Cochlear electrode array was partially inserted via round window. Sound field hearing threshold improvement was achieved.<br />

Mean tonal threshold was 46.2 dB HL. The patient could only detect environmental sounds and human voice but cannot<br />

discriminate vowels, words nor do sentences at 2 years of follow-up.<br />

Conclusion: Cochlear implantation is a feasible auditory restoration option in NF2 when cochlear anatomic and functional<br />

nerve preservation is achieved. The RLA is adequate for this purpose and features as an option for hearing preservation in NF2<br />

patients.<br />

Keywords: Neurofibromatosis 2; Cochlear Implantation; Hearing Loss.<br />

INTRODUCTION<br />

Neurofibromatosis type 2 (NF2) is an autosomal<br />

dominant disease consequent to mutation of both alleles of<br />

a suppressor tumor gene in the long arm of chromosome<br />

22 (1,2). Bilateral vestibular schwannoma is typical, occurring<br />

in 90% of the gene carriers (1). Progressive sensorineural<br />

hearing loss is almost certain, accompanied or not with<br />

dizziness and tinnitus.<br />

The first attempt at auditory restoration in a patient<br />

with NF2 and profound hearing loss was conducted by<br />

House and Hitselberger in 1979 (3). Auditory brainstem<br />

implant (ABI) has been the standard surgical treatment for<br />

these patients, to recover some degree of auditory capability<br />

and to enhance lip reading. In the 1990s, the promising<br />

idea of using a cochlear implant for auditory restoration<br />

became feasible. Auditory results of cochlear implantation<br />

are far better than those reported after ABI (4).<br />

The aim of this study is to describe a ase of a NF2-<br />

deafened-patient who underwent to vestibular schwannoma<br />

resection via retrolabyrinthine approach, with cochlear<br />

nerve preservation and cochlear implantation through the<br />

round window, at the same surgical time.<br />

CASE REPORT<br />

A 36-year-old woman with NF2 presented with a<br />

complaint of bilateral progressive sensorineural hearing<br />

loss for 10 years, which was similar in both ears. In 2009,<br />

the hearing loss became severe and communication<br />

problems aggravated. In 2006 she had undergone to<br />

partial tumor resection for facial nerve preservation on<br />

the left side at another institution. After surgery, the<br />

treatment was complemented with gama-knife radiation.<br />

She scheduled an appointment with our team seeking for<br />

a treatment for hearing rehabilitation. The tumor on her<br />

right side measured 1.5 cm (Figure 1).<br />

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Bento et al.<br />

Due to the size of the tumor we believed it would<br />

be more convenient to attempt hearing preservation in<br />

the right ear and to try a cochlear implant. After counseling<br />

she agreed with and accepted surgery. In March 2010, the<br />

patient underwent retrolabyrinthine approach with complete<br />

tumor resection and anatomic preservation of the<br />

cochlear and facial nerves.<br />

The retrolabyrinthine approach was performed<br />

with the patient placed in the classic supine position with<br />

the head rotated away from the surgeon. A retroauricular<br />

incision was made three centimeters posterior to the<br />

auricular sulcus extending to the mastoid tip. A muscle<br />

flap was placed anteriorly, allowing for the posterior<br />

retraction of the periosteum in order to drill the place into<br />

which the receiver/stimulator was lodged. A wild<br />

mastoidectomy was performed, exposing the entire posterior<br />

fossa dura mater, the sigmoid sinus and the jugular<br />

bulb. The posterior semicircular canal was partially drilled<br />

and obliterated, and the meatal plane was skeletonized.<br />

The vestibule was not opened. The posterior dura was<br />

opened from the base of the IAM to the anterior edge of<br />

the jugular bulb, extending to the edge of the sigmoid<br />

sinus. After cisternal drainage of cerebral spinal fluid the<br />

meatal plane was longitudinally opened and the tumor<br />

removed (Figure 2 A).<br />

The functional assessment of the cochlear nerve<br />

was evaluated through electrical promontory stimulation,<br />

which demonstrated present responses with poor<br />

morphology an unreliable reproducibility. Subsequently,<br />

cochlear implantation of a Nucleus 24RE Contour Advance<br />

(Cochlear Ltd., Lane Cove, Australia) through the round<br />

window was performed. The electrode array was partially<br />

inserted (6 active electrodes out) (Figures 2 B, 3).<br />

Impedances of electrodes measurements were adequate.<br />

The patient had absent intraoperative neural response<br />

telemetry (NRT) in Electrodes 1, 22, 11, 6 and 16 with<br />

25μs (Figure 4 A) and also in electrodes 11 and 15 with<br />

50μs (Figure 4 B). No complications or additional deficts<br />

occured.<br />

Figure 1. Coronal magnetic resonance imaging of the temporal<br />

bone showing bilateral cerebellopontine angle tumors<br />

with homogeneous enhancement after gadolinium injection<br />

on T1-weighted sequence, measuring 1.5 cm on the right side<br />

and 3.0 cm on the left side.<br />

Figure 2.View of the retrolabyrinthine approach with vestibular schwannoma being removed (arrow) (A). View<br />

of the posterior tympanotomy approach with electrode array inserted in the coclea via round windown (arrow<br />

head) (B). (MFD, middle fossa dura-mater; PFD, posterior fossa dura-mater; PSC, posterior semi-circular canal;<br />

LSC, lateral semi-circular canal).<br />

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Figure 3. High-resolution coronal (A) and axial (B) CT scans of the temporal bones showing insertion of the electrode array<br />

into the middle and basal turns of the cochlea.<br />

The cochlear implant was activated four weeks<br />

after surgery, with a Freedom speech processor with<br />

hearing sensation in 5 electrodes with advanced<br />

combination encoders (ACE) speech coding strategy<br />

900Hz of stimulation rate and 5 maxima, with 75μs pulse<br />

width. All other electrodes elicited visible facial nerve<br />

stimulation. Two months after activation she was referred<br />

to auditory training with a speech pathologist. With the<br />

increase in pulse width (150μs), 11 electrodes elicited<br />

auditory response with very little or no facial stimulation.<br />

Sound field hearing threshold improvement was achieved<br />

(Figure 5). The mean tonal threshold was 46.2 dBHL.<br />

Despite the auditory training and the improvement in<br />

hearing thresholds, the patient could only detect<br />

environmental sounds and human voice but cannot<br />

discriminate vowels, words nor do sentences at 2 years of<br />

follow-up.<br />

Figure 4. Intra-operative NRT showing absent evoked potentials<br />

on electrodes 1, 22, 11, 6, and 16 at 25-μs pulse width (A) and<br />

on electrodes 11 and 15 at 50-μs pulse width (B).<br />

DISCUSSION<br />

Cueva et al. (5) elicited auditory sensation through<br />

electrical promontory stimulation in 6 patients with NF2<br />

who underwent tumor removal with cochlear nerve<br />

preservation. Since then, about 31 cases of cochlear<br />

implantation in NF2 patients have been reported in the<br />

literature (6-16). The timing of implantation varied from<br />

simultaneously to 10 years after tumor resection.<br />

Regardless of the time of cochlear implantation or the<br />

approach used, these studies have reported a diverse<br />

range of speech perception outcomes, from no benefit to<br />

significantly better speech discrimination (14).<br />

Figure 5. Sound field hearing threshold before and 48 months<br />

after surgery with the cochlear implant in the right ear.<br />

The previously described approaches for CI in<br />

these patients were translabyrinthine, retrosigmoid or<br />

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Bento et al.<br />

middle cranial fossa. The translabyrinthine approach for<br />

schwannoma removal is a highly destructive method, and<br />

degenerative changes in the cochlea such as fibrosis or<br />

ossification are more pronounced and occur in a short<br />

time (13). Thus, we hypothesized that the RLA with<br />

simultaneous cochlear implantation could bring better<br />

results.<br />

Cochlear implantation offers advantages over ABI<br />

as reliable tonotopic stimulation of the auditory system.<br />

It is also less technically demanding and reduces the risk<br />

of major complications (16). As an attempt to avoid the<br />

overall poor hearing results of ABI and to limit the<br />

adverse effects of direct brainstem stimulation, cochlear<br />

implantation has been proposed and successfully<br />

performed in this case.<br />

Surgical tumor resection may result in mechanical<br />

or thermal injury to the cochlear nerve or labyrinthine<br />

artery and traction on the eighth nerve may cause<br />

avulsion of delicate auditory fibers at the habenula perforata<br />

(16). Some studies demonstrate that hearing preservation<br />

is achieved in only 32%-67% of cases (16). Nevertheless,<br />

more than 80% of such patients are left with an anatomically<br />

intact auditory nerve (16). Thus, although anatomic<br />

conservation of the cochlear nerve is fundamental, if the<br />

functional preservation is not achieved, the auditory<br />

results may be poor. The use of electrical promontory<br />

stimulation testing is controversial as positivity does not<br />

guarantee cochlear implant performance. Our patient´s<br />

test showed present responses and cochlear implant was<br />

performed with improvement in hearing thresholds, but<br />

she cannot discriminate speech.<br />

CONCLUSION<br />

Cochlear implantation is a feasible auditory restoration<br />

option in NF2 when cochlear anatomic and functional<br />

nerve preservation is achieved. The RLA approach was<br />

adequate for this purpose and presents an option for<br />

hearing preservation in NF2 patients.<br />

REFERENCES<br />

1. Kanowitz SJ, Shapiro WH, Golfinos JG, Cohen NL, Roland<br />

JT Jr. Auditory brainstem implantation in patients with<br />

neurofibromatosis type 2. Laryngoscope. 2004;114:2135-<br />

46.<br />

2. Vincenti V, Pasanisi E, Guida M, Di Trapani G, Sanna M.<br />

Hearing rehabilitation in neurofibromatosis type 2 patients:<br />

cochlear versus auditory brainstem implantation. Audiol<br />

Neurootol. 2008;13:273-80.<br />

3. Hitselberger WE, House WF, Edgerton BJ, Whitaker S.<br />

Cochlear nucleus implants. Otolaryngol Head Neck Surg.<br />

1984;92(1):52–4.<br />

4. Tran Ba Huy P, Kania R, Frachet B, Poncet C, Legac MS.<br />

Auditoryrehabilitation with cochlear implantation in patients<br />

with neurofibromatosis type 2.Acta Otolaryngol.<br />

2009;129(9):971–5.<br />

5. Cueva RA, Thedinger BA, Harris JP, Glasscock ME 3rd.<br />

Electrical promontory stimulation in patients with intact<br />

cochlear nerve and anacusis following acoustic neuroma<br />

surgery. Laryngoscope. 1992;102:1220–24.<br />

6. Tono T, Ushisako Y, Morimitsu T: Cochlear implantation<br />

in an intralabyrinthine acoustic neuroma patient after<br />

resection of an intracanalicular tumor. J Laryngol Otol.<br />

1996;110:570–73.<br />

7. Graham J, Lynch C, Weber B, Stollwerck L, Wei J, Brookes<br />

G: The magnetless Clarion cochlear implant in a patient<br />

with neurofibromatosis 2. J Laryngol Otol. 1999;113:458–<br />

63.<br />

8. Temple RH, Axon PR, Ramsden RT, Keles N, Deger K,<br />

Yucel E: Auditory rehabilitation in neurofibromatosis type<br />

2: a case for cochlear implantation. J Laryngol Otol.<br />

1999;113:161–63 .<br />

9. Ahsan S, Telischi F, Hodges A, Balkany T: Cochlear<br />

implantation concurrent with translabyrinthine<br />

acoustic neuroma resection. Laryngoscope.<br />

2003;113:472–74.<br />

10. Nolle C, Todt I, Basta D, Unterberg A, Mautner VF, Ernst<br />

A: Cochlear implantation after tumor resection in<br />

neurofibromatosis type 2: impact of infra- and postoperative<br />

neural response telemetry monitoring. ORL. 2003;65:230–<br />

34.<br />

11. Aristegui M, Denia A: Simultaneous cochlear implantation<br />

and translabyrinthine removal of vestibular schwannoma in<br />

an only hearing ear: report of two cases (neurofibromatosis<br />

type 2 and unilateral vestibular schwannoma). Otol Neurotol.<br />

2005;26:205–10.<br />

12. Lustig LR, Yeagle J, Driscoll CL, Blevins N, Francis H,<br />

Niparko JK. Cochlear implantation in patients with<br />

neurofibromatosis type 2 and bilateral vestibular<br />

schwannoma. Otol Neurotol. 2006;27:512-18.<br />

13. Vincenti V, Pasanisi E, Guida M, Di Trapani G, Sanna M.<br />

Hearing rehabilitation in neurofibromatosis type 2 patients:<br />

cochlear versus auditory brainstem implantation. Audiol<br />

Neurootol. 2008;13:273-80.<br />

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Bento et al.<br />

14. Celis-Aguilar E, Lassaletta L, Gavilán J. Cochlear<br />

implantationin patients with neurofibromatosis type 2 and<br />

patients with vestibular schwannoma in the only hearing<br />

ear. Int J Otolaryngol. 2012;2012:157497.<br />

16. Carlson ML, Breen JT, Driscoll CL, Link MJ, Neff BA, Gifford<br />

RH, Beatty CW. Cochlear implantation in patients with<br />

neurofibromatosis type 2: variables affecting auditory<br />

performance. Otol Neurotol. 2012;33(5):853-62.<br />

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tumor removal?Otol Neurotol. 2001;22(4):497–500.<br />

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355


COURSE SCHEDULE 2013<br />

SET UP AND MAKE YOUR ENTRY<br />

JULY<br />

DATE<br />

III COCHLEAR IMPLANT COURSE FOR EDUCATIONAL PROFESSIONALS 5<br />

ENDOSCOPIC ENDONASAL SURGERY COURSE - UFBA 18,19,20<br />

IV ADVANCED COURSE IN OTOLOGY AND NEUROTOLOGY ANATOMY (JOÃO PESSOA) 25,26,27<br />

AUGUST<br />

DATE<br />

XIII UNIVERSITY EXTENSION COURSE - SEMESTER 2 07,21,28<br />

V OTOLARYNGOLOGY ANATOMY COURSE 3<br />

11 TH STOMATOLOGY JOURNEY 02, 03<br />

OTOMASTER 05,12,19,26<br />

VIII AUDITORY EVOKED POTENTIALS COURSE: THEORETICS AND PRACTICE 02, 03<br />

TINNITUS TRAINING COURSE 23<br />

VII OTONEUROLOGY BASIC COURSE - PRINCIPLES AND PRACTICE 26,27<br />

VII ELECTRONISTAGMOGRAPHY INTERPRETATION COURSE: THEORETICS AND PRACTICE 28,29,30<br />

SEPTEMBER<br />

DATE<br />

OTOMASTER 02,09,16,23<br />

XIII UNIVERSITY EXTENSION COURSE - SEMESTER 2 04,11,18,25<br />

II NOSE - LIVE ENDOSCOPIC ENDONASAL SURGERY 18,19<br />

101º TEMPORAL BONE DISSECTION COURSE (SP) 24,25,26,27<br />

X COURSE OF SWALLOWING DISORDERS AND LARYNGOPHARYNGEAL REFLUX - THEORETICS AND<br />

PRACTICE<br />

05, 06<br />

XXIII OTORHINOLARYNGOLOGY JOURNEY OF FEDERAL UNIVERSITY OF MINAS GERAIS AND<br />

VIII OTORHINOLARYNGOLOGY JOURNEY OF FORL-MG<br />

13,14<br />

IV POLYSOMNOGRAPHIC REPORT INTERPRETATION COURSE 11<br />

XV COURSE OF SNORING AND OBSTRUCTIVE SLEEP APNEA - THEORETICS AND PRACTICE 12,13<br />

OCTOBER<br />

DATE<br />

OTOMASTER 7,28<br />

III BROADCASTED COURSE IN RADIOLOGY IN OTORHINOLARYNGOLOGY 21,22,24,25<br />

XIII UNIVERSITY EXTENSION COURSE - SEMESTER 2 09,16,23, 30<br />

ENDOSCOPY COURSE FOR OTORHINOLARYNGOLOGISTS - THEORETICS AND PRACTICE - STEPS 1 AND 2 09,10,11<br />

ENDOSCOPIC ENDONASAL SURGERY COURSE - UFBA <strong>17</strong>,18,19<br />

LARYNX MICROSURGERY COURSE WITH DISSECTION - THEORETICS AND PRACTICE <strong>17</strong>,18,19<br />

X COURSE OF REHABILITATORS OF COCHLEAR IMPLANT USERS OF HCFMUSP 04, 05<br />

IX MULTICENTER COCHLEAR IMPLANT COURSE BASIC MODULE - USP/SP, BAURU AND FEDERAL UNIVERSITY<br />

OF PARANÁ<br />

24,25,26<br />

Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. xxx-xxx, Jul/Aug/September - 2013.<br />

356


COURSE SCHEDULE 2013<br />

SET UP AND MAKE YOUR ENTRY<br />

NOVEMBER<br />

DATE<br />

OTOMASTER 04,11,18,25<br />

IV FACIAL NERVE COURSE - LECTURES AND DISSECTION PRACTICE 25,26,27<br />

XIII UNIVERSITY EXTENSION COURSE - SEMESTER 2 06,13,27<br />

III MEETING WITH PARENTS OF CHILDREN WITH HEARING LOSS AND AUDITORY IMPLANTS TEAM OF<br />

HCFMUSP<br />

9<br />

DECEMBER<br />

DATE<br />

OTOMASTER 02,09,16<br />

102º TEMPORAL BONE DISSECTION COURSE (SP) 10,11,12,13<br />

MAGNA CONFERENCE CLOSING 11<br />

Visit the website of FORL www.forl.<strong>org</strong>.br and make your application.<br />

Promotion and Organization:<br />

Teodoro Sampaio Street, 483<br />

Pinheiros - São Paulo / Brazil<br />

tel.: +55 11 3068-9855<br />

e-mail: cursosforl@forl.<strong>org</strong>.br<br />

357www.forl.<strong>org</strong>.br


358


congressoforl1@forl.<strong>org</strong>.br<br />

Phone: (55-11) 3081 4223<br />

http://www.forl.<strong>org</strong>.br/congresso2013<br />

359


www.internationalarchivesent.<strong>org</strong><br />

360

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