Revista Arquivos 17(3) - ingles.pmd - Internationalarchivesent.org
Revista Arquivos 17(3) - ingles.pmd - Internationalarchivesent.org
Revista Arquivos 17(3) - ingles.pmd - Internationalarchivesent.org
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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 />
Address for correspondence:<br />
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Design: Vagner Bertaco Severino<br />
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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Vilkman, E. A survey on the occupational safety and<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 />
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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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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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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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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 />
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3. Golding-Kushner KJ, et al. Standartization for the reporting<br />
of nasopharyngoscopy and multiview videofluoroscopy: a<br />
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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 />
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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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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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GP, Roithmann R, et al. Hearing impairment and<br />
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18. ANSI. Maximum Permissible Ambient Noise Levels for<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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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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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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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 />
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3. Mannino DM, Holguin F. Epidemiology and global impact<br />
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4. Campos HS. O preço da DPOC. Pulmão RJ. 2003;12(4):<br />
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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 />
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2001 FTRP by ASHA]. [cited 2007 Mar 3]. Available from:<br />
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7. Michou E, Hamdy S. Cortical input in control of<br />
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2009;<strong>17</strong>(3):166-71.<br />
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Chronic Obstructive Lung Disease: Global strategy for the<br />
diagnosis, management and prevention of chronic<br />
obstructive pulmonary disease: GOLD executive summary.<br />
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9. Wedzicha JA, Seemungal TA. COPDexacerbations:<br />
defining their cause and prevention. Lancet.<br />
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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 />
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12. Marik P. Aspiration pneumonitis and aspiration<br />
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14. Crary M, Mann G, Groher M. Initial psychometric<br />
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JL. A penetration aspiration scale. Dysphagia.<br />
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<strong>17</strong>. Menezes AM, Victora CG, Rigatto M. Prevalence and<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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279
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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[Curitiba]: Universidade Federal do Paraná; 2005. 111 p.<br />
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8. Woo P, Noordzij JP. Glottal area waveform analysis of<br />
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12. Lehmann W, Pampurik J, Guyot JP. Laryngeal pathologies<br />
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14. Eckley CA, Swensson J, Duprat AC, Donati F, Costa HO.<br />
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Otorrinolaringol. 2008:74(4):508-11.<br />
15. Neves BM, Neto JG, Pontes P. Diferenciação<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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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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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 />
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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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303
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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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 />
Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.<strong>17</strong>, n.3, p. 315-320, Jul/Aug/September - 2013.<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 />
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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 />
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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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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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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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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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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 />
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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 />
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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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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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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 />
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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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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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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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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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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 />
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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 />
15. Belal A. Is cochlear implantation possible after acoustic<br />
tumor removal?Otol Neurotol. 2001;22(4):497–500.<br />
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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 />
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OCTOBER<br />
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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 />
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NOVEMBER<br />
DATE<br />
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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 />
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