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Yıl/Year:2022 Cilt/Volume11 Sayı/Isue:2

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RESPIRATORYCASEREPORTS

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Respir Case Rep 2022;11(2): I-III

EDITÖRDEN

EDITORIAL

Completing the 10th Year in Respiratory Case

Reports

Respiratory Case Reports Dergisinde 10. Yılı Tamamlarken…

RESPIRATORY CASE REPORTS

“Case Reports” are the oldest known type of

scientific publication in history. We have

compeleted 10 years in our publication life, which

we started by saying “Preparing case reports for

young academicians is a very important step in

learning how to write scientific manuscript” (1).

The Respiratory Case Reports is an international,

peer-reviewed, quick-refereeing open access

journal published online by LookUs, İstanbul,

Türkiye. The journal entered international indexes

by performing a first in the category “Case

Reports” two years after its publication. Respiratory

Case Reports is indexed by EBSCO, DOAJ,

EMBASE, Index Copernicus, Academic Index,

Turkish Citation Index, and TUBITAK/ULAKBIM

Medical Database. The inaugural issue was

released in June 2012, and in 2022, we published

the nearly 500 papers. A complete breakdown of

the articles by years and by journal section can be

seen in Figure 1 and Figure 2, respectively.The

majorty of these manuscripts have been case

reports. Submissions are authentic, understandable,

educational and clinically interesting to an

international audience of respiratory physicians,

trainees and researchers in all respiratory

subspecialties, as well as clinicians in related fields

(Figure 3). It has published all issues in a timely

manner without delay up to this moment. From

modest beginnings, registered readers of the

Journal have increased year by year to over

134,130 readers in June 2022. “Respiratory Case

Reports” journal has reached you with the diligent

effort of the LookUs team with the understanding of

“Unconditional Support to Education”. We wanted

our journal to be freely accessible by all our

colleagues.

We would like to thank the 780 authors who sent

manuscripts to our journal and our valued

reviewers who carefully evaluated these

mansucripts. We would also like to express our

gratitude to our proffessors on the editorial board

and our publication manager Tuba Avcılar, PhD

and her team.

Figure 1: Total articles published by year

Figure 2: Total articles published by journal section (n=465)

Figure 3: Distribution of accepted/published articles by categories

I

Respiratory Case Reports

You can find our reviewers who evaluated most

manuscripts (Table 1), the manuscripts which are cited

most (Table 2), and the most dowloaded manuscripts

(Table 3) in this appendix, which we make short breakdown

of the last ten years.

Our journal has come to this day with the contributions of

our esteemed colleagues.

We hope that it will continue to grow and develop for many

decades.

Regards,

Zafer Kartaloğlu, Oğuzhan Okutan

Department of Pulmonology, Health Science University, Sultan

II. Abdülhamid Han Training and Research Hospital, İstanbul,

Türkiye

Correspondence: Zafer Kartaloğlu, Department of Pulmonology,

Health Science University, Sultan II. Abdülhamid Han Training and

Research Hospital, İstanbul, Türkiye

zkartaloglu@hotmail.com

REFERENCES

1- Kartaloğlu Z, Okutan O. Importance case reports in

medical literature. Respir Case Rep 2012; 1:1. [CrossRef]

Table 1: Top Ten Reviewers who evaluated articles

Rank Reviewer Name Surname

1 Aydanur EKICI

2 Tayfun ÇALIŞKAN

3 Levent DALAR

4 Ahmet Emin ERBAYCU

5 Attila SAYGI

6 Arzu ERTÜRK

7 Ersin DEMIRER

8 Adem GÜNGÖR

9 Akif TURNA

10 Mustafa ERELEL

Table 2: Ten Most Cited Respiratory Case Reports Articles Since June 2012

Rank Reference Title Issue, Year DOI

1 Ünlü M, Çimen P, Arı G, A Successfully Treated Severe Case of Extensively Vol. 4, No. 3 10.5505/respircase.2015.32932

Dereli MŞ.

Drug-Resistant Tuberculosis During Pregnancy (2015)

2 Tanz R, Elalami I, Azami MA,

Allaoui M, Errihani H,

Ichou M.

Advanced primary pulmonary carcinosarcoma: a case

report and review of the literature

Vol. 6, No. 2

(2017)

10.5505/respircase.2017.35220

3 Sezen CB, Çelik A,

Akboğa SA, Akyürek N,

Taştepe Aİ.

Intrapulmonary solitary fibrous tumor Vol. 3, No. 3

(2013)

10.5505/respircase.2014.44154

4 Demirer E, Ghattas C,

Rahman HA, Elamin E.

Current Management of Hypothermia: From Theory

to Application

Vol. 1, No. 2

(2012)

10.5505/respircase.2012.36844

5 Oruç M, Meteroğlu F,

Ahmet Elbey A, Şahin A,

Monis S.

A Case of Agressive Fibromatosis in the Chest Wall Vol. 4, No. 1

(2015)

10.5505/respircase.2015.92005

6 Borelli EP. Maybe it is More than Pneumonia: Case Report of an

Intralobar Sequestration in a 20-Year-Old Male

Vol. 6, No. 2

(2017)

10.5505/respircase.2017.92499

7 Ediboğlu Ö, Kıraklı SC,

Tatar D, Tuksavul FF.

A case of ARDS due to Legionella Pneumonia treated

with ECMO

Vol. 3, No. 3

(2014)

10.5505/respircase.2014.60252

8 Yazgan S, Yoldaş B,

Gürsoy S.

Video-Assisted Thoracoscopic Removal of a Mysterious

Foreign Body Causing Pneumothorax

Vol. 7, No. 2

(2018)

10.5505/respircase.2018.04900

9 Çörtük M, Tanrıverdi E,

Yıldırım BZ, Abbaslı K,

Özgül MA, Çetinkaya E.

A Case of Foreign Body Aspiration Diagnosed in Early

Adulthood

Vol. 5, No. 2

(2016)

10.5505/respircase.2016.92063

10 Kafadar C, Öztürk E,

Kara K, Sağlam M, Tutar S.

Pleural Lipoma: a Case Report Vol. 4, No. 3

(2015)

10.5505/respircase.2015.69885

Cilt - Vol. 11 Sayı - No. 2

II

Completing the 10th Year in Respiratory Case Reports | Kartaloğlu and Okutan

Table 3: Ten Most Frequently Viewed Respiratory Case Reports Articles Since Inaugural Issue in June 2012

Rank Reference Title Issue, Year DOI

1 Kartaloğlu Z, Okutan O. What to consider when preparing a case Vol. 1, No. 2 10.5505/respircase.2012.29591

report

(2012)

2 Taş D, Demirer E, Çiftçi F,

Okutan O, Kartaloğlu Z.

Lipoid Pneumonia Caused by Diesel Fuel

Aspiration: A Case Report

Vol. 1, No. 1

(2012)

10.5505/respircase.2012.02996

3 Yıldız Gülhan P, Ekici A,

Bulcun E, Ekici MS.

Pulmonary Langerhans cell histiocytosis X: an

analysis of four cases

Vol. 2, No. 3

(2013)

10.5505/respircase.2013.70298

4 Çelik B, Yılmaz MA,

Pirzirenli MG, Şahin M.

Granulomatous Diseases Should be Considered

in Mediastinal Lymphadenopathies with

High F-18 FDG Uptake on PET-CT Scans

Vol. 6, No. 2

(2017)

10.5505/respircase.2017.98705

5 Yıldız Aİ, Özkurt S, Kıter G,

Alaçam Z, Aydoğan BE.

Lung Toxicity Due To Mesalamine Vol. 2, No. 1

(2013)

10.5505/respircase.2013.43531

6 Çimen D, Ekici M, Bulcun E,

Ekici A.

BOOP (Bronchiolitis Obliterans Organizing

Pneumonia) and Renal Amyloidosis Secondary

to Infected Cystic Bronchiectasi

Vol. 2, No. 1

(2013)

10.5505/respircase.2013.19480

7 Darılmaz Yüce G,

Sarınç Ulaşlı S.

Chronic Cough Due to Tracheal Diverticulum Vol. 1, No. 2

(2012)

10.5505/respircase.2012.54264

8 Ahat Çimen D, Ekici A,

Bulcun E, Ekici M.

Delayed Diagnosis in a Patient with Bronchiectasis:

Swyer-James-MacLeod Syndrome

Vol. 2, No. 1

(2013)

10.5505/respircase.2013.14622

9 Okutan O, Ayten Ö, Demirer

E, Ugan N, Kartaloğlu Z.

Pulmonary carcinoid tumor presenting with

widespread bone metastasis

Vol. 1, No. 2

(2012)

10.5505/respircase.2012.87597

10 Akbaş A, Seyhan E C, Sökücü

S N, Altın S, Günlüoğlu G,

Altay S.

Systemic Nocardiosis In A Diabetic Patient Vol. 1, No. 2

(2012)

10.5505/respircase.2012.92485

III

www.respircase.com

Respir Case Rep 2022;11(2):55-58 DOI: 10.5505/respircase.2022.70437

OLGU SUNUMU

CASE REPORT

Robotic-assisted Bronchoscopy with

Endobronchial Ultrasound for the Diagnosis a

of Peripheral Lung Nodule with

Lymphadenopathy: A Case Report

Lenfadenopatili Periferik Akciğer Nodülünün Tanısında Endobronşiyal

Ultrason ile Robotik Yardımlı Bronkoskopi: Olgu Sunumu

Sai Priyanka Pulipaka, Katherine Walsh, Alejandra Yu Lee-Mateus, Daniel Hernandez,

Rocio Castillo-Larios, David Abia-Trujillo, Sebastian Fernandez-Bussy


Abstract

Some 95% of all detected lung nodules are benign,

although it is essential to determine the underlying

cause, given that lung cancer is the leading cause of

oncological death in the United States. When choosing

a diagnostic tool, reducing the number of procedures

without compromising the diagnostic value is

essential. This case report focuses on the successful

use of the latest robotic-assisted bronchoscopy in

combination with existing radial and linear endobronchial

ultrasound-guided transbronchial needle

aspiration for the biopsy of a peripherally located

lung nodule and mediastinal lymph nodes. Combining

the two procedures allowed the peripheral nodule

and lymph nodes to be sampled with no complications

in a single procedure. The nodule showed no

cancerous growth, although the lymph nodes showed

granulomas consistent with Histoplasma.

Key words: Peripheral lung nodule, Robotic-assisted

bronchoscopy, Endobronchial ultrasound.

Öz

Akciğer nodüllerinin yaklaşık %95'i iyi huylu olmakla

beraber, ABD'de onkolojik ölümlerin önde gelen en

sık nedeni akciğer kanseri olduğu için akciğer nodüllerin

türünü belirlemek gerekmektedir. Bir teşhis aracı

seçerken, teşhis değerinden ödün vermeden, yapılan

işlem sayısını azaltabilmek önemlidir. Bu olgu sunumunda,

periferik yerleşimli bir akciğer nodülü ve

mediastinal lenf nodlarının biyopsisinde mevcut radyal

ve lineer endobronşiyal ultrason rehberliğinde

transbronşiyal iğne aspirasyonu ile beraber, robotik

yardımlı bronkoskopinin başarılı bir şekilde birlikte

kullanılması anlatılmaktadır. Tek bir işlem ile hem

periferik nodül hem de lenf nodülü, komplikasyonsuz

olarak örneklendi. Periferik nodülde malingnite yoktu,

ancak lenf nodülü Histoplazma ile uyumlu bulundu.

Anahtar Sözcükler: Periferik akciğer nodülü, Robotik

yardımlı bronkoskopi, Endobronşiyal ultrason.

Department of Pulmonary, Allergy, and Sleep Medicine, Mayo

Clinic, Jacksonville, Florida

Pulmoner, Alerji ve Uyku Tıbbı Departmanı, Mayo Clinic,

Jacksonville, Florida

Submitted (Başvuru tarihi): 07.12.2021 Accepted (Kabul tarihi): 31.02.2022

Correspondence (İletişim): Sai Priyanka Pulipaka, Department of Pulmonary, Allergy, and Sleep Medicine, Mayo Clinic, Jacksonville,

Florida

e-mail: pulipaka.priyanka@mayo.edu

55


Despite the continued advances in procedures, the diagnosis

of peripheral lung nodules (PLN) remains challenging.

Computerized Tomography-guided Transthoracic

Needle Biopsy (CT-TTNB) has been the standard approach

for many years, with diagnostic value of over 90%

(1). The simultaneous sampling of lymph nodes for mediastinal

staging is not possible with CT-TTNB, and there is

a higher rate of pneumothorax than with biopsies done

through bronchoscopy (2,3). The diagnostic yield of PLN

through standard navigation bronchoscopy is 70%, which

is low when compared to CT-TTNB (2,4). We report a

here on a case of PLN with associated lymphadenopathy

diagnosed as Histoplasmosis via robotic-assisted bronchoscopy

(RAB) in combination with EBUS-TTNA. RAB

allowed the difficult peripheral regions of lungs to be

reached, while EBUS allowed the sampling of the lymph

nodes in the mediastinum in a single procedure (5).

CASE

A 67-year-old female with post liver-transplant immunosuppression

and with a past medical history of chronic

kidney disease, squamous cell carcinoma of the helix in

the left ear, hepatocellular carcinoma (HCC), diastolic

heart failure, obstructive sleep apnea and pulmonary

hypertension, presented for a routine checkup. A chest

Computerized Tomography (C.T.) for HCC surveillance

revealed a 9mm nodule in the subpleural lateral basal

right lower lobe along the inferior aspect of the right

major fissure (Figure 1). Other than a mild early-morning

nonproductive cough, she referred to no other symptoms,

recent travel or exposure to environmental agents. Tests

for Histoplasma antigen, Blastomyces and coccidioidomycosis

were negative. Due to the peripheral location

and suspicion for malignancy, we used RAB and radial

and linear EBUS to sample the nodule and mediastinal

lymph nodes (Figure 2), without result. The sample from

the nodule confirmed no cancerous growth. The biopsy

results of one of the lymph nodes with GMS stain showed

non-necrotizing granulomas, morphologically suggestive

of yeast cell histoplasmosis (Figure 3). Bronchoalveolar

lavage revealed one colony of a filamentous fungus. The

patient was started on Voriconazole rather than Itraconazole

due to her past diastolic heart failure. At 2-week

follow-up, the patient was found to be tolerating the

treatment well with no reported complications. The most

recent CT revealed no considerable change in the size of

the nodule (Figure 4).

DISCUSSION

Peripherally located lung nodules smaller than 10mm are

often difficult to diagnose, but can be diagnosed by CT-

TTNA or bronchoscopy-guided biopsy. CT-TTNA has a

reported sensitivity of 90% and specificity of 97% in the

diagnosis of lung cancer, although complications such as

pneumothorax (25%), bleeding, infection and the risk of

seeding the tumor are common (2). CT-TTNA prevents

the staging of mediastinal lymph nodes, which is crucial

for the diagnosis of lung cancer. When CT-TTNA is used,

a second procedure is increased screenings result in more

incidentally found lung nodules; it is essential to balance

the potential harm from unnecessary procedures with the

benefits of early diagnosis. In recent studies, RAB has

been used for the diagnosis of actinomycosis, Loeffler

syndrome and mycobacteria (2).

The lymph node biopsy I the present study yielded positive

for Histoplasma – a dimorphic fungus causing a granulomatous

infection that is endemic to the central and

eastern parts of the United States, including Ohio and

Mississippi, as well as South America, Africa, Asia and

Australia. The infection can have a variable presentation,

ranging from asymptomatic to severe disseminated disease.

Disseminated infections are common in immunosuppressed

patients. Diagnostic tests such as antigen

detection and serology are routinely used, but can be

non-diagnostic in the presence of pulmonary nodules.

Fine needle biopsy or surgical resection is used for a

definitive diagnosis. Nodules can closely resemble a malignant

lesion on C.T. and show increased uptake on PET

scans. There are a few reports of Histoplasma being misdiagnosed

as cancer, as the giant cells in Histoplasma

can be mistaken for malignant lymphoblastic cells. Itraconazole,

Voriconazole and fluconazole are the preferred

treatment approaches for mild to moderate infections,

and amphotericin B for severe conditions.

Figure 1: The red circled region represents the peripheral lung nodule in

the right lung

Cilt - Vol. 11 Sayı - No. 2 56

Robotic-assisted Bronchoscopy with Endobronchial Ultrasound for the Diagnosis a of Peripheral Lung Nodule with Lymphadenopathy: A Case Report |

Pulipaka et al.

Figure 2: Image from the robotic-assisted bronchoscopy. The red box

shows the distance of the scope from the target nodule, the yellow box

shows the scope distance from the pleura, the green box is a 3D image

of the lung nodule

lesion, although more studies are needed in this regard

(1,5).

In our case, with the patient's previous cancer history, a

newly diagnosed 9mm lung nodule raised a high suspicion

for malignancy, although other infectious causes,

however, could not be ruled out due to the patient’s immunosuppressive

status. Recent studies of robotic-assisted

bronchoscopy have reported a diagnostic value of 90% in

the diagnosis of peripherally located nodules, with a

3.6% risk of pneumothorax and 2.8% risk of bleeding.

(2,5). Radial EBUS with RAB allowed us to visualize the

peripherally located lung nodule before sampling, and

linear EBUS-TBNA allowed the real-time sampling of

mediastinal lymph nodes. Using RAB and EBUS, we accessed

the peripheral nodules and the mediastinal and

hilar lymph nodes during the same procedure, avoiding

multiple procedures.

Figure 3: The marked area shows granuloma consistent with histoplasmosis

CONCLUSION

The challenges faced in the management of peripherally

located lung nodules can be overcome through a comprehensive

approach involving newer modalities in conjunction

with more established approaches. Combining

RAB with EBUS-TBNA can help in enhancing diagnostic

yield, avoiding multiple procedures with reducing the

potential for complications in the diagnosis of PLN. The

approach has provided promising results in the diagnosis

of infections that present with lung nodules, such as Histoplasma,

which can be mistaken for malignancy.

CONFLICTS OF INTEREST

None declared.

Figure 4: The circled area shows a nodule of the same size

RAB has the potential to be used in the future as a therapeutic

tool for the delivery of ablative treatments, for the

treatment of inoperable tumors or metastatic lesions with

photodynamic therapy, and for microwave ablation, radiofrequency

ablation and cryoablation. RAB could also

be used to deliver direct anti-microbial drugs into the

AUTHOR CONTRIBUTIONS

Concept - S.P.P., K.W., A.Y.L., S.F., D.H., R.C., D.A.;

Planning and Design - S.P.P., K.W., A.Y.L., S.F., D.H.,

R.C., D.A.; Supervision - S.P.P., K.W., A.Y.L., S.F., D.H.,

R.C., D.A.; Funding - S.F., D.A.; Materials - S.P.P., K.W.,

S.F.; Data Collection and/or Processing - S.P.P.; Analysis

and/or Interpretation - S.P.P.; Literature Review - S.P.P.,

K.W., A.Y.L., S.F., D.H., R.C., D.A.; Writing - S.P.P., K.W.;

Critical Review - S.P.P., K.W., A.Y.L., S.F., D.H., R.C.,

D.A.

YAZAR KATKILARI

Fikir - S.P.P., K.W., A.Y.L., S.F., D.H., R.C., D.A.; Tasarım

ve Dizayn - S.P.P., K.W., A.Y.L., S.F., D.H., R.C., D.A.;

Denetleme - S.P.P., K.W., A.Y.L., S.F., D.H., R.C., D.A.;

Kaynaklar - S.F., D.A.; Malzemeler - S.P.P., K.W., S.F.;

57 www.respircase.com


Veri Toplama ve/veya İşleme - S.P.P.; Analiz ve/veya

Yorum - S.P.P.; Literatür Taraması - S.P.P., K.W., A.Y.L.,

S.F., D.H., R.C., D.A.; Yazıyı Yazan - S.P.P., K.W.; Eleştirel

İnceleme - S.P.P., K.W., A.Y.L., S.F., D.H., R.C., D.A.

REFERENCES

1. Agrawal A, Hogarth DK, Murgu S. Robotic bronchoscopy

for pulmonary lesions: a review of existing technologies

and clinical data. J Thorac Dis 2020; 12:3279-86.

[CrossRef]

2. Khan T, Usman Y, Abdo T, Chaudry F, Keddissi JI,

Youness HA. Diagnosis and management of peripheral

lung nodule. Ann Transl Med 2019; 7:348. [CrossRef]

3. DiBardino DM, Yarmus LB, Semaan RW. Transthoracic

needle biopsy of the lung. J Thorac Dis 2015; 7(Suppl

4):S304-S16. [CrossRef]

4. Chen YB, Jiang JH, Mao JY, Huang JA. Diagnostic value

of endobronchial ultrasound-guided transbronchial needle

aspiration (EBUS-TBNA) in solitary mediastinal, hilar

lymphadenectasis, or peribronchial lesions: Six cases reports

and review of literature. Medicine (Baltimore) 2016;

95:e5249. [CrossRef]

5. Chaddha U, Kovacs SP, Manley C, Hogarth DK, Cumbo-

Nacheli G, Bhavani SV, et al. Robot-assisted bronchoscopy

for pulmonary lesion diagnosis: results from the initial

multicenter experience. BMC Pulm Med 2019; 19:24.

[CrossRef]



OLGU SUNUMU

CASE REPORT

Coexistence of Sarcoidosis and Silicosis:

Case Series

Sarkoidoz ve Silikozis Birlikteliği: Olgu Serisi

Melike Yüksel Yavuz,

Yucel Demiral


Abstract

The differential diagnosis of silicosis includes sarcoidosis,

berylliosis, hypersensitivity pneumonia,

malignancy, tuberculosis and other granulomatous

infections. Sarcoidosis is a systemic granulomatous

disease with an unknown etiology, although it is

thought that occupational exposure to such substances

as silica and beryllium may be a trigger. An incomplete

occupational history may lead to a diagnosis

of sarcoidosis rather than pneumoconiosis in

many cases, although various associations of these

two diseases, such as the coexistence and detection

of one before or after the other, have been reported

in literature. The coexistence of sarcoidosis and silicosis

is discussed in the present study with reference

to five cases who applied to the Occupational Diseases

department of a university hospital. Active

pulmonary tuberculosis and lung malignancy were

excluded in all cases, and histopathologic examinations

of all samples were reported as non-caseating

granulomatous inflammation. In the specimen of one

case, a birefringent body was identified upon polarized

microscopy. In the light of these cases, it is

aimed to draw attention to the usefulness of a wellreceived

occupational history in the differential diagnosis

and etiology of sarcoidosis and silicosis.

Key words: Sarcoidosis, Silicosis, differential diagnosis.

Öz

Silikozisin ayırıcı tanısı sarkoidoz, berilyoz, hipersensitivite

pnömonisi, malignite, tüberküloz ve diğer granülomatöz

enfeksiyonları içerir. Sarkoidoz etiyolojisi

tam olarak bilinmeyen sistemik granülomatöz bir

hastalıktır ve silika, berilyum gibi mesleki maruziyet

faktörlerin tetikleyici ajan olarak rol oynayabileceği

düşünülmektedir. Eksik ve dikkatsiz bir meslek öyküsü

birçok olguda pnömokonyoz yerine sarkoidoz tanısına

yol açabilir. Ek olarak bu iki hastalığın bir arada

görülmesine veya bir diğerinin ötekinden önce saptaması

gibi durumlara da çeşitli makalelerde dikkat

çekilmiştir. Bu yazıda bir üniversite hastanesinin iş ve

meslek hastalıkları bölümüne başvuran beş olgu

üzerinden sarkoidoz ve silikoz birlikteliği tartışılmıştır.

Tüm olgularda aktif akciğer tüberkülozu ve akciğer

malignitesi dışlandı. Tüm olguların biyopsi materyallerinin

histopatolojik incelemeleri kazeifiye olmayan

granülomatöz inflamasyon olarak rapor edildi. Bir

olguda polarize mikroskopta ışığı çift kıran cisim

gösterildi. Burada, bu olgular ışığında iyi alınmış bir

meslek öyküsünün sarkoidoz ve silikozisin ayırıcı tanısı

ve etiyolojisini saptamadaki faydasına dikkat çekmek

amaçlanmıştır.

Anahtar Sözcükler: Sarkoidoz, Silikoz, ayırıcı tanı.

Department of Work and Occupational Diseases, Dokuz Eylül

University Faculty of Medicine, İzmir, Türkiye

Dokuz Eylül Üniversitesi Tıp Fakültesi, İş ve Meslek Hastalıkları

Bilim Dalı, İzmir


Correspondence (İletişim): Melike Yüksel Yavuz, Department of Work and Occupational Diseases, Dokuz Eylül University Faculty of

Medicine, İzmir, Türkiye

e-mail: yukselmelike@windowslive.com

59


The differential diagnosis of silicosis includes sarcoidosis,

berylliosis, hypersensitivity pneumonia, malignancy, tuberculosis

and other granulomatous infections. Sarcoidosis

is a systemic granulomatous disease of unknown etiology,

although it is thought that occupational exposure to

such substances as silica and beryllium may play a role,

as well as infectious agents and genetic factors, as triggering

agents. It is known that the interaction of silica

particles with macrophage activation and IL-1, TNF, fibronectin,

fibrogenic cytokine and free radicals leads to

immunoactivation and fibrogenesis. The presence of

similar clinical, laboratory, radiological and histopathological

findings of silicosis and sarcoidosis may be challenging

for clinicians in a differential diagnosis, and an

incomplete and inattentive occupational history may lead

to a diagnosis of sarcoidosis rather than pneumoconiosis

in many cases (1). Furthermore, various associations of

these two diseases, such as the coexistence and detection

of one before or after the other have been reported on in

literature. In the present study, the coexistence of sarcoidosis

and silicosis is discussed with reference to five

cases who applied to the Occupational Diseases Department

of a university hospital.

images of all cases revealed mediastinal lymph nodes

associated with lung parenchymal nodules (Figure 1 and

2). While the radiological findings were stable at 3 and 4

years of follow-up in case-2 and case-3, whose final

diagnosis was pneumoconiosis, mild progression was

detected in case-1 in the third year of follow-up. Since the

diagnoses of silicosis and sarcoidosis were new in case-4

and case-5, respectively, no specific follow-up could not

be performed.

The demographics and clinical features of our cases are

presented in Table-1, while their business lines and occupational

dust exposures are presented in Table-2.

CASE

A total of five cases are described, all of whom are male,

aged 27–51 years. Three of the cases were diagnosed

initially with sarcoidosis and treated accordingly, in which

sarcoidosis was diagnosed at the earliest in the first year

and at the latest in the third year following admission.

One case had been diagnosed with silicosis 6 years before

their diagnosis of sarcoidosis.

Lymph node biopsies were performed by flexible bronchoscopy

(FOB) in case-1, mediastinoscopy in case-2,

both peripheral lymph node excisional biopsy and FOB in

case-3, peripheral lymph node excisional biopsy in case-

4 and by endobronchial ultrasonography (EBUS) in case-

5. The histopathologic examinations of all samples reported

non-caseating granulomatous inflammation. In

the specimen of case 4, a birefringent body was revealed

in a polarized microscopy.

Active pulmonary tuberculosis and lung malignancy were

excluded in all cases. A fiberoptic bronchoscopy was

performed in all cases, and the bronchial washing cytology

results were found to be benign, and there was no

reproduction of tuberculosis bacillus. Since the lymphocyte

proliferation test could not be measured, sensitivity to

exposures could not be investigated. Computed tomography

(CT)/high resolution computed tomography (HRCT)

Figure 1: Thorax radiological images of cases 1, 2 and 3. (a) Thorax

HRCT of Case 1, millimetric nodules and subcarinal lymphadenomegaly

in bilateral lung parenchyma; (b) Thorax CT of Case 2, millimetric nodules

and subcarinal lymphadenomegaly in bilateral lung parenchyma; (c)

Thorax CT of Case 3, Right lung upper apical localized multiple nodules

with radial borders, the largest reaching 2 cm in diameter, and subcarinal,

hilar calcified lymphadenomegaly

DISCUSSION

Of the five cases presented in this study, four were diagnosed

initially with sarcoidosis, and silicosis was considered

during follow-ups. One case previously diagnosed

with silicosis was later confirmed to have sarcoidosis as

well. Accurate occupational history taking is crucial for

the differential diagnosis of silicosis in patients with sarcoidosis

and for the detection of the etiology. As stated by

Seaton, an incomplete history may result in a diagnosis of

sarcoidosis rather than silicosis in the same patient (1).


Coexistence of Sarcoidosis and Silicosis: Case Series | Yüksel Yavuz et al.

Table 1: Demographics and clinical features of the cases

Cases Age ILO Evaluation Thorax HRCT or CT Spirometry Sarcoidosis Features

Case 1 27 Quality 2, 2/2,

untreated follow-up

p/p ax

* 20- 25mm mediastinal lymphademomegaly

* Milimetric nodules observed more

intensely in the upper zones of the

bilateral parenchyma

* Bilateral upper lobe apex and

posterior peribronchial thickenings

FEV1: 3.13 ml %87

FVC: 3,50 ml %84

FEV1/FVC: %89

Diagnosis Times

* Diagnosis of

sarcoidosis 3 years

before admission

* Diagnosis of silicosis

on admission

Case 2

51

Quality 2,

1/1,p/s hi

* Largest subcarinal 2 cm, hilar

lymphadenomegaly

* Nodular pleural thickenings in

bilateral upper lobes Right lung

upper lobe 9 mm calcific granuloma

* Millimetric nodule at the level of

the right lung major fissure

FEV1: 2.57 ml %81

FVC: 3,36 ml %85

FEV1/FVC: %77

untreated follow-up

* Diagnosis of

sarcoidosis 2 years

before admission


on admission

Case 3

49

Quality 1, q/p

2/2 ax

* Lymphadenomegaly, the largest

13 mm, in the right axillary region

* Bilateral upper lobe apical and

lower lobe medial 2 cm multiple

nodules with radial borders

* Consolidation of right lung middle

lobe laterally and left lung upper

lobe anterior

* Right pleural calcific focus Left

hemidiaphragm elevated, left less

pleural fluid

FEV1: 1.70 ml %48

FVC: 2.05 ml %47

FEV1/FVC: %77

long term oxygen

therapy and

corticosteroid

therapy

* Diagnosis of

sarcoidosis 3 years

before admission


on admission

Case 4 33 Quality 2, q/q

2/2

A, hi, left pleural

thickening

* Multiple calcified lymph nodes in

the mediastinum, the largest of

which is 30*14 mm

* Millimetric multiple calcific nodules,

diffuse in bilateral upper lobe

and lower lobe superior, multiple

micronodular areas in bilateral

lower lobes

FEV1: 3.87 ml %87

FVC: 4.28ml %79

FEV1/FVC: 90.35

Methylprednisolone

4 mg, azathioprine

5 mg and hydroxychloroquine

200 mg for the last

1 year *

* After the diagnosis

of sarcoidosis, due

to joint pain and

Anti-ribosomal P

positivity, it was

diagnosed by the

rheumatology

department.

* Diagnosis of

sarcoidosis 3 years

before admission


on admission

Case 5 44 Quality 1, r/r,

3/3 hi, ax

* Mediastinal and hilar lymph

nodes, the largest of which is 20

mm in diameter in the subcarinal

region

* Multiple nodules with miliary

distribution pattern in all lobes,

bilaterally in the upper lobes, focal

consolidations with local merging of

nodules defined in the upper lobes

and anterior segments

FEV1:12.07 ml

%58,5

FVC: 2.67 ml %62

FEV1/FVC: %77.6

corticosteroid

therapy

Serum ANA was

weakly positive, and

serum ACE level

was 114.2 IU/L

* Diagnosis of sarcoidosis

within the

year of admission

* Diagnosed with

silicosis 6 years ago

ILO: İnternational Labour Organization, HRCT: High Resolution Computed Tomography, CT: High Resolution Computed Tomography,

FEV1: Forced Expiratory Volume In 1 Second, FVC: Forced Vital Capacity

In a case-control study including 3,663 cases and 7,326

controls, it was stated that occupational silica exposure

increased the incidence of sarcoidosis in men aged 20–

65 years, and sarcoidosis was reported at a higher rate in

those who had been exposed to silica in the last 5 years

of their occupation (2). The cases presented in the pre-



sent study had an average of 13.5 years of exposure to

silica in various employment areas, and there was an

average of 4.8 years (2–11 years) between the time of

admission and the most recent exposure.

The prevalence of sarcoidosis has been shown to be

higher in workers exposed to silica. In a retrospective

cohort study conducted in 2017, a study of the medical

records of workers in 10 iron foundries revealed that

silica exposure increased the risk of sarcoidosis (SIR 3.94;

95% CI 1.07 to 10.08) (3). In the aftermath of the attacks

against the World Trade Center, it was found that the

incidence of sarcoidosis or sarcoidosis-like granulomatous

lung disease increased in the 5-year observation

period of the firefighters working in search and rescue

teams when compared to the results of examinations

carried out 15 years earlier (4). In a study from Sweden in

2019, 371 cases of sarcoidosis were identified among

297,917 male workers, and it was shown that smokers

had an increased risk of developing sarcoidosis when

exposed to high levels of silica dust when compared to

non-smokers (5). In another case series, silica exposure

was identified in six of eight sarcoidosis cases (6), while a

study examining death records in the United States between

1988 and 1999, 3,393 of 7,118,535 recorded

deaths were identified as being sarcoidosis-related and

sarcoidosis was given as the underlying cause of death in

1,579 of these cases. It has also been reported that the

risk of sarcoidosis mortality related to occupational exposure

differs for those of different sexes and races (7).

Table 2: Work places and occupational dust exposures of the cases

Cases

Case 1

Case 2

Case 3

Job/Work

place

Dental

technician

Ceramic

Cement

Rubber

dough

production

Task

Metal casting

Metal leveling

Sandblasting

Porcelain polishing

Skeleton making

Press-mill operatör

Machine maintainer

Production and

Slaughter

Case 4 Mining Physical analysis -

Elimination operator

Exposure

Silica

Plaster

Wax

Resin

Silica

Silica

Rubber

Sulfur

Kaulen

Kuartz

Case 5 Foundry Sandcore making Silica

Resin

Time of

Exposure

14 years

21 years

14 years

9 years

8 years

Figure 2: Thorax radiological images of cases 4 and 5. (a) Thorax CT of

Case 4, millimetric nodules up to 2 cm in diameter and subranial, hilar

calcified lymphadenomegaly in the right lung lobe superior; (b) Thorax

CT of Case 5, millimetric multiple noduleswith miliary distribution pattern

in both lungs, predominantly in the upper lobes, in all lobes and accompanied

by subpleural involvement, multiple mediastinal lymphadenomegaly;

(c) 6 years ago Thorax CT of Case 5, millimetric nodules and

subcranial lymphadenomegaly in bilateral lung parenchyma

In the cases in the present study, the coexistence of sarcoidosis

and silicosis, or sequential associations of the

two, can occur. In a case report, a 45-year-old man who

had worked in metal molding and had been exposed to

silica presented with symptoms for 3 years. The reported

asteroid bodies, giant cells and granulomatous inflammation

in the histopathological evaluation of an open lung

biopsy were compatible with sarcoidosis, and silica particles

were also found in the nodules (8). Roegel et al. (9)

reported a case of rapidly progressive radiological findings

of pseudotumoral silicosis during multivisceral exacerbation

with iritis and erythema nodosum in a miner with

pulmonary and nodal sarcoidosis treated with corticosteroids.

The patient was biopsied by thoracotomy, and sarcoid

granulomas with silicotic masses were found limited

to the upper lobe, along with sarcoid granulomas in the

middle lobe and hilar lymph nodes, and the patient was

thus diagnosed with sarcoido-silicosis. The authors concluded

that sarcoidosis promoted the rapid development

of silicosis due to the changes in immunity, and was unexpected

since the exposure had been moderate. They

went on to speculate that this may be due to the deficiency

in the elimination of silica particles from the lungs.


Coexistence of Sarcoidosis and Silicosis: Case Series | Yüksel Yavuz et al.

Clinician may not be able to give a definitive diagnosis of

silicosis or sarcoidosis, but may diagnose patients with

sarco-silicosis, silico-sarcoidosis or sarcoid-like granulomatous

disease of the lung related to silica. The treatment

modality of the patient may, therefore, change depending

on which diagnosis is made. The “from treatment

to diagnosis” method is frequently resorted to in

these cases. A 67-year-old male patient who had been

exposed to silica in a cement factory presented with bilateral

centrilobular nodules, hilar and mediastinal lymphadenopathy

with calcification on Thorax CT, and was

diagnosed with suspected sarcoidosis. Upon the detection

of birefringent material under polarized light microscopy

in a mediastinal lymph node, EBUS and transbronchial

needle biopsy were performed, and a diagnosis of silicosis-related

sarcoid-like granulomatous lung disease was

made instead of sarcoidosis. After the replacement of the

treatment modality with corticosteroid and azathioprine

combination therapy, the patient’s radiological findings

and clinical progress improved (10). Beijer et al. (11)

reported on a 49-year-old patient who had been working

as a plasterer in construction for 30 years. The patient

was diagnosed with silicosis, and demonstrated silica

sensitivity in a lymphocyte proliferation sensitivity test,

along with birefringent material on a polarization microscopic

examination of an open lung biopsy. His symptoms,

however, persisted and fibrosis due to silicosis continued,

even under prednisone and azathioprine treatment.

Symptomatic and radiological improvement was

detected in the 7th month after the patient was started on

infliximab, and so his condition was rediagnosed as sarcoidosis.

In the present study, two of the cases were treated

for sarcoidosis and the other three were followed up

without treatment. Furthermore, two of our patients who

were treated for sarcoidosis experienced radiological

progression and were recently rediagnosed with silicosis.

There are some limitations to the present study. The lack

of lymphocyte proliferation tests in our cases was the

missing aspect in terms of showing sensitivity. Furthermore,

the level of exposure was limited to what the subjects

reported in their occupational histories, and there

were no personal dust exposure measurements. If large

tissue samples of the lung parenchyma were available,

our prediction would be better supported. In addition, the

lack of a standard follow-up approach to the cases was

also a shortcoming.

Silica can activate the autoimmune mechanism and lead

to the emergence of autoantibodies, and so it has been

associated with SLE, scleroderma, rheumatoid arthritis

and sarcoidosis (12). The role of silica as a trigger of

sarcoidosis has been recognized (13), although it has not

yet been clarified whether inorganic dusts can trigger

sarcoidosis in those with and without genetic predispositions.

In 2021, an article entitled “Sarcoidosis: An Occupational

Disease?’” noted that silica exposure can be

associated with sarcoidosis in various sectors, such as

agriculture, construction, fire brigade, foundries, timber

and mining (14). In the light of the cases presented in the

present study, we suggest that a well-documented occupational

history, including exposure levels, may be helpful

in the differential diagnosis of sarcoidosis and silicosis,

and the etiology of parenchymal lung disease.


None declared.


Concept - M.Y.Y., Y.D.; Planning and Design - M.Y.Y.,

Y.D.; Supervision - M.Y.Y., Y.D.; Funding - M.Y.Y.; Materials

- M.Y.Y.; Data Collection and/or Processing -

M.Y.Y.; Analysis and/or Interpretation - M.Y.Y.; Literature

Review - M.Y.Y.; Writing - M.Y.Y.; Critical Review -

M.Y.Y., Y.D.

YAZAR KATKILARI

Fikir - M.Y.Y., Y.D.; Tasarım ve Dizayn - M.Y.Y., Y.D.;

Denetleme - M.Y.Y., Y.D.; Kaynaklar - M.Y.Y.; Malzemeler

- M.Y.Y.; Veri Toplama ve/veya İşleme - M.Y.Y.; Analiz

ve/veya Yorum - M.Y.Y.; Literatür Taraması - M.Y.Y.;

Yazıyı Yazan - M.Y.Y.; Eleştirel İnceleme - M.Y.Y., Y.D.

REFERENCES

1. Seaton A. Silica dust and sarcoidosis. Occup Med (Lond)

2020; 70:139. [CrossRef]

2. Graff P, Larsson J, Bryngelsson IL, Wiebert P, Vihlborg P.

Sarcoidosis and silica dust exposure among men in

Sweden: a case-control study. BMJ Open 2020;


3. Vihlborg P, Bryngelsson IL, Andersson L, Graff P. Risk of

sarcoidosis and seropositive rheumatoid arthritis from occupational

silica exposure in Swedish iron foundries: a

retrospective cohort study. BMJ Open 2017; 7:e016839.

[CrossRef]

4. Crowley LE, Herbert R, Moline JM, Wallenstein S, Shukla

G, Schechter C, et al. "Sarcoid like" granulomatous pulmonary

disease in World Trade Center disaster responders.

Am J Ind Med 2011; 54:175-84. [CrossRef]



5. Jonsson E, Järvholm B, Andersson M. Silica dust and sarcoidosis

in Swedish construction workers. Occup Med

(Lond) 2019; 69:482-6. [CrossRef]

6. Rafnsson V, Ingimarsson O, Hjalmarsson I, Gunnarsdottir

H. Association between exposure to crystalline silica and

risk of sarcoidosis. Occup Environ Med 1998; 55:657-

60. [CrossRef]

7. Liu H, Patel D, Welch AM, Wilson C, Mroz MM, Li L, et al.

Association Between Occupational Exposures and Sarcoidosis:

An Analysis From Death Certificates in the United

States, 1988-1999. Chest 2016; 150:289-98. [CrossRef]

8. Tousheed S, Sen T, Kumar H, Bv M. Silicosis and Sarcoidosis:

A Rare Association. Chest 2014; 146, 421A.

[CrossRef]

9. Roegel E, Pauli G, Dechoux J, Warter A, Le Bouffant L,

Martin JC, et al. Silicosis with a rapid and pseudotumoral

course occurring in a case of pre-existant progressive

and treated pulmonary sarcoidosis in a miner little

exposed to silicosis risk: sarcoido-silicosis (author's transl).

Poumon Coeur 1981; 37:195-202.

10. Mochizuka Y, Kono M, Katsumata M, Hirama R, Watanuki

M, Oshima Y, et al. Sarcoid-like granulomatous

lung disease with subacute progression in silicosis. Intern

Med 2022; 61:395-400. [CrossRef]

11. Beijer E, Meek B, Kromhout H, van Es HW, Seldenrijk K,

Drent M, et al. Sarcoidosis in a patient clinically diagnosed

with silicosis; is silica associated sarcoidosis a new

phenotype?. Respir Med Case Rep 2019; 28:100906.

[CrossRef]

12. Pollard KM. Silica, silicosis, and autoimmunity. Front Immunol

2016; 7:97. [CrossRef]

13. Grunewald J, Spagnolo P, Wahlström J, Eklund A. Immunogenetics

of disease-causing inflammation in sarcoidosis.

Clin Rev Allergy Immunol 2015; 49:19-35.

[CrossRef]

14. Oliver LC, Zarnke AM. Sarcoidosis: an occupational disease?

Chest 2021; 160:1360-7. [CrossRef]



OLGU SUNUMU

CASE REPORT

Munchausen's Syndrome as a Cause of

Hemoptysis in a Prisoner

Bir Mahkumda Hemoptizi Nedeni Olarak Munchausen Sendromu

Hulya Dirol 1 , Fatma Deniz 1 , Yaşar Gülnur Güdül 2


Abstract

Factitious hemoptysis is fairly rare condition, the

diagnosis of which can be challenging to physicians.

A 40-year-old incarcerated male with hemoptysis for

four years had previously undergone a detailed investigation,

but the etiology of the hemoptysis could not

be determined. The hemoptysis has increased in

frequency and quantity, however, numerous diagnostic

tests, including radiological imaging and bronchoscopy,

has been unable to reveal the localization

and etiology of the bleeding. As we were about to

diagnose idiopathic hemoptysis, some suspicious

behaviors and contradictory statements led us to

conduct a psychiatric evaluation, and the patient was

subsequently diagnosed with axis two personality

disorder and factitious disorder. Factitious hemoptysis

is difficult to diagnose and may be confused with

idiopathic hemoptysis, and should be considered in

patients with hemoptysis in which the location and

cause of bleeding cannot be determined after a detailed

examination.

Key words: Munchausen's syndrome, Factitious hemoptysis,

Factitious disorder.

1 Department of Chest Diseases, Akdeniz University Faculty of

Medicine, Antalya, Türkiye

2 Akdeniz University Faculty of Medicine, Antalya, Türkiye

Öz

Yapay hemoptizi oldukça nadirdir ve tanısı doktorları

zorlar. Kırk yaşındaki erkek mahkûmun dört yıldır

hemoptizisi vardı. Daha önce detaylı inceleme yapılmış

ancak hemoptizinin etiyolojisi belirlenememişti.

Son günlerde hemoptizi sıklığı ve miktarı artmıştı. Biz

de radyolojik görüntüleme ve bronkoskopi dahil

birçok tanısal tetkikler yaptık ancak kanamanın lokalizasyonunu

ve etiyolojisini belirleyemedik. Tam idyopatik

hemoptizi teşhisi koyacakken bazı şüpheli davranışlar

ve çelişkili ifadeler gördük. Psikiyatrik değerlendirmenin

ardından hastaya ikinci eksen kişilik

bozukluğu ve yapay bozukluk tanısı konuldu. Yapay

hemoptizinin teşhisi zordur ve idiyopatik hemoptizi ile

karıştırılabilir. Detaylı bir muayene ile kanama yeri ve

nedeni belirlenemeyen hemoptizi hastalarında düşünülmelidir.

Anahtar Sözcükler: Munchausen sendromu, Yapay

hemoptizi, Yapay bozukluk.

1 Akdeniz Üniversitesi Tıp Fakültesi Göğüs Hastalıkları Anabilim

Dalı, Antalya

2 Akdeniz Üniversitesi Tıp Fakültesi, Antalya


Correspondence (İletişim): Hulya Dirol, Department of Chest Diseases, Akdeniz University Faculty of Medicine, Antalya, Türkiye

e-mail: hulyadirol@akdeniz.edu.tr

65


Factitious hemoptysis is an uncommon form of Munchausen's

syndrome, which refers to the imitation of many

well-known diseases (1). It is a syndrome that should be

considered by physicians, as it can often lead to urgent/unnecessary

surgical operations or unnecessary

investigations for diagnosis. Such patients can deftly mimic

an acute illness and convince the physician for the

presence of the disease. Cases with Munchausen's syndrome

rarely present with hemoptysis. The patients mostly

have an underlying psychiatric disease (2). For the incarcerated,

factitious hemoptysis may be the only means of

prison release (3). Here, we present a patient with hemoptysis

who, despite many investigations, was not identified

with any pathological findings and was eventually

diagnosed with factitious disorder.

CASE

A 40-year-old incarcerated male presented to the emergency

department with a complaint of hemoptysis that

had started 4 years ago and increased in frequency and

quantity. He complained of nonpleuretic pain, which he

considered to be a sign of hemoptysis, but no other respiratory

symptoms. He had been incarcerated for 5 years

and had previously been investigated in a university hospital

for hemoptysis, but the etiology could not be identified.

A pulmonary artery embolization was recommended,

but the patient declined. Today, the patient experiences

hemoptysis from time to time in larger quantities, expectorating

one tea-glass of fresh blood a day.

The patient was stable hemodynamically in the emergency

room. He claimed to be allergic to contrasting agents

and refused pulmonary computerized tomography (CT)

angiography. A pulmonary perfusion and ventilation

scintigraphy revealed no thromboembolism. There was no

pathology in high resolution lung tomography (Figure 1),

and no pathological finding in an upper respiratory tract

examination. An abdominal examination was normal,

while a slight elevation was noted in liver function tests.

There was no significant finding on ultrasound and viral

etiological markers were negative. The patient had a

positive occult fecal blood test, but refused a rectal examination.

No active bleeding was observed on endoscopy

or colonoscopy. He refused bronchoscopy, and claimed

to be allergic to anesthetic agents. We referred the patient

to the Allergy-Immunology Department, where he

was found to be allergic to neither anesthetic drugs nor

contrast agents. A Pulmonary CT Angiography revealed

no vascular abnormality or pulmonary thromboembolism.

We performed bronchoscopy under conscious sedation,

but found no lesion or bleeding in the tracheobronchial

tree. An evaluation of a bronchus biopsy revealed mild

chronic inflammation. For the evaluation of bleeding

disorders, we examined Von Willebrand Factor, Protein S,

Protein C, fibrinogen and activated Protein C Resistance

levels, carried out a Platelet Function Test and assessed

the coagulation parameters, all of which were normal.

During the follow up in the hospital, the patient had fever

and hematuria. A blood culture revealed Stenotrophomonas

maltophilia and Pseudomonas alcaligenes, and

he was placed on wide spectrum antibiotic treatment. A

urine analysis revealed erythrocytes, but no crescent formation.

Renal Doppler ultrasonography and a serum

assay for vasculitis and connective tissue disease were

normal. The patient had undergone a Lung CT angiography

before, but there was no mention of any anaphylactic

reaction to contrast, all of which aroused suspicion

of factitious hemoptysis. After close follow-up, we found

needles hidden by the patient in his personal locker. He

admitted aspirating blood through intravenous needles

and presenting the blood as if it had come from the oral

and urethral routes. After a psychiatric evaluation, he was

diagnosed with axis two personality disorders and factitious

disorder, and referred to the high-security Psychiatry

service.

DISCUSSION

Hemoptysis, or the expectoration of blood, is a significant

pulmonary symptom that may occur with different etiological

factors. The source of the blood may be the airways,

lung or the pulmonary vasculature. Many different laboratory

and imaging investigations are required for the

determination of the etiology. In the majority of patients

the etiology of hemoptysis can be identified (4), although

diagnosis may sometimes be harder, especially, if the

patient is a good imitator and hides his/her real intentions,

or is s/he has a factitious disorder.

Figure 1: Normal high-resolution chest computerized tomography


Munchausen's Syndrome as a Cause of Hemoptysis in a Prisoner | Dirol et al.

Munchausen's syndrome is a factitious disorder that was

first described by Asher in 1951 (5). In factitious disorders,

patients deliberately mimic a disease by producing physical

or psychological symptoms, and Munchausen's syndrome

is a more severe and chronic form of this disorder.

Most reported cases of Munchausen's syndrome are in

the pediatric age group (6). The most common clinical

problems reproduced intentionally include abscess, pain,

hypoglycemia, anemia, bleeding, rashes, seizures, dizziness,

bleaching, vomiting, diarrhea and fever. Bleeding

symptoms in Munchausen's syndrome usually present as

hemoptysis, hematemesis, hematochezia, vaginal bleeding,

hematuria or ecchymosis (7). Munchausen's syndrome

is a disease that is difficult to diagnose, and can

easily be overlooked. A series of examinations should be

performed to exclude a real reason for hemoptysis. The

clinical and laboratory findings of the patients are generally

inconsistent, and for a diagnosis of Munchausen's

syndrome, organic pathologies should be excluded. Uzuner

et al. (8) claims that diagnosis requires the physician

to be suspicious and to adopt a multidisciplinary approach.

Since the hemoptysis in our patient was accompanied by

hematuria, we suspected pulmonary-renal syndromes,

however, the chest CT of the patient was incompatible

with diffuse alveolar hemorrhage, and as there was no

nephritic sediment in urinalysis or rapidly progressive

renal function loss, we excluded pulmonary-renal syndrome.

Moreover, the serum assay for vasculitis and

connective tissue disease was normal. We also investigated

parenchymal pathologies of the lung via high resolution

chest CT and pulmonary thromboembolism via lung

CT angiography, none of which revealed any pathological

finding. Furthermore, a bronchoscopy for the evaluation

of the airways was conducted, but failed to identify

the site of any bleeding or to reveal the etiology. As we

were about to diagnose idiopathic hemoptysis, we recognized

some suspicious behaviors and contradictory statements,

and found that he had hidden a needle in his

locker, after which he admitted producing the bleeding

consciously.

In conclusion, factitious hemoptysis produced voluntarily

and consciously by a patient in a symptom that can be

imitated (9). Although it is mostly associated with an underlying

psychiatric illness, it can sometimes be used for

legal or economic gain (10). The diagnosis of factitious

hemoptysis can be challenging for physicians, and can be

mistaken for idiopathic hemoptysis. High clinical suspicion

is required for diagnosis, and it should definitely be

considered in patients with hemoptysis whose bleeding

localization and the cause cannot be determined, even

after a detailed investigation. Patients should be closely

observed for suspicious behaviors and evaluated for psychiatric

disease, as efforts to make a diagnosis may lead

to many noninvasive tests and invasive procedures performed

to exclude possible etiologies.


None declared.


Concept - H.D., F.D., Y.G.G.; Planning and Design -

H.D., F.D., Y.G.G.; Supervision - H.D., F.D., Y.G.G.;

Funding - H.D.; Materials - H.D.; Data Collection and/or

Processing - H.D.; Analysis and/or Interpretation - H.D.;

Literature Review - H.D., F.D.; Writing - H.D.; Critical

Review - H.D., F.D., Y.G.G.

YAZAR KATKILARI

Fikir - H.D., F.D., Y.G.G.; Tasarım ve Dizayn - H.D., F.D.,

Y.G.G.; Denetleme - H.D., F.D., Y.G.G.; Kaynaklar -

H.D.; Malzemeler - H.D.; Veri Toplama ve/veya İşleme -

H.D.; Analiz ve/veya Yorum - H.D.; Literatür Taraması -

H.D., F.D.; Yazıyı Yazan - H.D.; Eleştirel İnceleme - H.D.,

F.D., Y.G.G.

REFERENCES

1. Turner J, Reid S. Munchausen's syndrome. Lancet 2002;

359:346-9. [CrossRef]

2. Weber B, Gokarakonda SB, Doyle MQ. Munchausen

Syndrome. In: StatPearls. Treasure Island (FL): StatPearls

Publishing; 2021.

3. Sinha A, Smolik T. Striving to die: Medical, legal, and

ethical dilemmas behind factitious disorder. Cureus 2021;


4. Tsoumakidou M, Chrysofakis G, Tsiligianni I, Maltezakis

G, Siafakas NM, Tzanakis N. A prospective analysis of

184 hemoptysis cases: diagnostic impact of chest X-ray,

computed tomography, bronchoscopy. Respiration 2006;

73:808-14. [CrossRef]

5. Asher R. Munchausen's syndrome. Lancet 1951; 1:339-

41. [CrossRef]

6. Sheridan MS. The deceit continues: an updated literature

review of Munchausen syndrome by proxy. Child Abuse

Negl 2003; 27:431-51. [CrossRef]



7. Shaw RJ, Dayal S, Hartman JK, DeMaso DR. Factitious

disorders by proxy: pediatric condition falsification. Harv

Rev Psyciatry 2008; 16:215-24. [CrossRef]

8. Uzuner S, Bahali K, Kurban S, Erenberk U, Cakir E. A

pediatric case of factitious disorder with unexplained

bleeding symptoms. Gen Hosp Psychiatry 2013;

35:679.e7-8. [CrossRef]

9. Baktari JB, Tashkin DP, Small GW. Factitious hemoptysis.

Adding to the differential diagnosis. Chest 1994;

105:943-5. [CrossRef]

10. Mucha SM, Varghese LA, French RE, Shade DA. Separating

fact from factitious hemoptysis: a case report. Crit

Care Nurse 2014; 34:36-42. [CrossRef]



OLGU SUNUMU

CASE REPORT

A Rare Cause of Respiratory Failure: Negative

Pressure Pulmonary Edema

Solunum Yetmezliğinin Nadir Bir Nedeni: Negatif Basınçlı Pulmoner Ödem

Başak Sayınalp 1 , Oğuz Abdullah Uyaroğlu 2


Abstract

We report here on the successful management of a

patient who developed dyspnea after an elective

arthroscopic procedure and who was diagnosed with

Negative Pressure Pulmonary Edema (NPPE). A 29-

year-old male patient with no known medical history

was admitted to our hospital due to a rotator cuff tear

sustained during a military operation. An arthroscopic

procedure under general anesthesia was performed;

however, the patient developed dyspnea 2 hours after

extubation. He was desaturated, tachypneic and

began to produce frothy pink sputum. Pulmonary

computed-tomography angiography revealed widespread

ground-glass opacities in both lungs suggesting

non-cardiogenic pulmonary edema without acute

pulmonary thromboembolism. The patient was thus

diagnosed with NPPE and treated with non-invasive

mechanical ventilation, intravenous furosemide and

inhaled short-acting beta-agonists. Significant recovery

was observed in a couple of days, and the patient

discharged after his respiratory symptoms abated.

NPPE should immediately be suspected in individuals

who develop respiratory failure following extubation,

as it can be life threatening.

Key words: Acute respiratory failure, negative pressure

pulmonary edema, non-cardiac pulmonary edema.

Öz

Bu olgu sunumunda, elektif artroskopik işlem sonrası

nefes darlığı gelişen, negatif basınçlı pulmoner ödem

(NBPÖ) tanısı konan ve başarıyla tedavi edilen bir

hasta sunulmuştur. Yirmi dokuz yaşında, herhangi bir

tıbbi öyküsü olmayan erkek hasta, askeri operasyon

sırasında gelişen rotator manşet yırtığı nedeniyle

hastanemize başvurdu. Genel anestezi altında artroskopik

işlem yapıldı. Ancak hastada ekstübasyondan 2

saat sonra dispne gelişti. Hastada satürasyon düşüklüğü

ve takipne ile birlikte ve köpüklü pembe balgam

görüldü. Bilgisayarlı tomografi pulmoner anjiyografide

tromboemboli saptanmayıp, her iki akciğerde

kardiyojenik olmayan pulmoner ödem düşündüren

yaygın buzlu cam opasiteleri saptandı. Hasta NBPÖ

tanısı konularak invazif olmayan mekanik ventilasyon,

intravenöz furosemid ve inhale kısa etkili betaagonistler

ile tedavi edildi. Birkaç gün içinde belirgin

bir iyileşme görülen hasta herhangi bir solunum

semptomu olmadan taburcu edildi. Ekstübasyon

sonrası solunum yetmezliği gelişen kişilerde hayatı

tehdit edici bir tablo olan NBPÖ akla gelmelidir.

Anahtar Sözcükler: Akut solunum yetmezliği, negatif

basınçlı pulmoner ödem, kardiyojenik olmayan pulmoner

ödem.

1 Department of Internal Medicine, Hacettepe University Faculty of

Medicine, Ankara, Türkiye

2 Department of General Internal Medicine, Hacettepe University

Faculty of Medicine, Ankara, Türkiye

1 Hacettepe Üniversitesi Tıp Fakültesi, İç Hastalıkları Anabilim

Dalı, Ankara

2 Hacettepe Üniversitesi Tıp Fakültesi, İç Hastalıkları Anabilim

Dalı, Genel Dahiliye Bilim Dalı, Ankara


Correspondence (İletişim): Başak Sayınalp, Department of Internal Medicine, Hacettepe University Faculty of Medicine, Ankara,

Türkiye

e-mail: bsayinalp@gmail.com

69


Negative pressure pulmonary edema (NPPE) is a rare

complication that can occur as a result of laryngospasm

following extubation after an operation requiring general

anesthesia (1). As a potentially life-threatening emergency

usually seen in young and healthy individuals, and that

can ultimately be recovered with a quick diagnosis, all

physicians should keep NPPE in mind during the postoperative

period as a clinically significant phenomenon.

Here we report on a patient who was managed successfully

after developing dyspnea following an elective arthroscopic

procedure and being diagnosed with NPPE.

CASE

A 29-year-old male soldier with no known medical history

was admitted to our hospital with a rotator cuff tear sustained

during a military operation. An arthroscopic procedure

under general anesthesia was performed, and the

tear was repaired. The patient developed dyspnea 2

hours after extubation, and was found to be desaturated

(SpO 2 at room air 75%) and tachypneic (respiratory rate:

26-28/min), and began to produce a frothy pink sputum.

The patient was immediately placed on 10 L/min oxygen

support with a mask. Bilateral coarse crackles were heard

during auscultation of the lungs, and a chest X-ray revealed

bilateral recently-developed pulmonary infiltrations

(Figure 1). Blood tests showed leukocytosis (predominantly

neutrophils), mild hypoxemia, lactic acidosis and elevated

D-dimer levels. The laboratory results are presented

in Table 1. It was found out that earlier in the operation

room the patient had developed laryngospasm following

extubation. As a result of the sudden onset dyspnea and

the elevated D-dimer levels, a pulmonary computedtomography

(CT) angiography for acute pulmonary

thromboembolism (PTE) was performed. No sign suggesting

acute PTE was observed, although widespread

ground-glass opacities were seen in both lungs, suggesting

non-cardiogenic pulmonary edema (Figure 2). The

Electrocardiogram showed normal sinus rhythm and did

not show any ischemic change. Transthoracic echocardiography

revealed normal left ventricular systolic function.

The patient was diagnosed with NPPE, and was administered

intravenous furosemide and inhaled short-acting

beta-agonists. The patient was transferred to the intensive

care unit where he received non-invasive mechanical

ventilation, and significant recovery was observed in a

couple of days. His dyspnea and sputum production regressed

entirely. A control chest X-ray is shown in Figure

1. The patient was discharged after his need for oxygen

support abated.

DISCUSSION

NPPE is a type of non-cardiogenic pulmonary edema,

and is a post-operative complication that can be lifethreatening

unless diagnosed and treated early. It is usually

seen in young and healthy individuals with a prevalence

of 0.05–0.1% (1), and is prevalent in those who are

prone to airway obstruction. Risk factors include obesity,

short and thick neck, obstructive sleep apnea syndrome,

oropharyngeal and head/neck surgeries (2). The duration

between airway obstruction and the development of pulmonary

edema is only a couple of minutes in many cases

reported to date (1).

NPPE occurs due to an increase in negative intrathoracic

pressure (greater than -100 cmH2O, which is approximately

ten times the normal negative intrathoracic pressure),

to upper airway obstructions and to the transfer of

fluid to the pulmonary interstitium. High intrathoracic

pressure results in increased venous return and elevated

capillary hydrostatic pressure. In addition, adrenergic

activation and increased pulmonary vascular resistance

caused by hypoxia and acidosis result in right ventricular

expansion, deviation of the interventricular septum towards

left ventricle and left ventricular diastolic dysfunction.

Increased venous return and elevated pulmonary

wedge pressure cause fluid to pass into the pulmonary

interstitium, and pulmonary edema develops (3).

Figure 1: Bilateral pulmonary infiltrations on chest X-ray during respiratory

distress (left), resolution of infiltrations after treatment (right)

Figure 2: Bilateral widespread ground glass opacities that are more

dominant posteriorly, and interstitial thickening (cobblestone pattern),

which indicates non-cardiogenic pulmonary edema


A Rare Cause of Respiratory Failure: Negative Pressure Pulmonary Edema | Sayınalp et al.

Table 1: Laboratory results of the patient at the time of respiratory failure

Parameter Value Normal Range

Hemoglobin (g/dL) 16,9 13-17

Leukocyte count (x103/μL) 24,8 4,3-10,3

Neutrophil count (x103/μL) 23,2 2,1-6,1

Thrombocyte count (x103/μL) 286 156-373

Creatinine (mg/dL) 1,02 0,67-1,17

AST (U/L) a 30 <50

ALT (U/L) b 37 <50

Sedimentation rate (mm/h) 2 0-20

CRP (mg/dL) c 0,36 0-0,5

Myoglobin (μg/L) 67,4 17,4-105,7

Troponin I (ng/L) 3,4 14-42,9

CK-MB (μg/L) d 2 0,6-6,3

D-dimer (mg/L) 8,22 0-0,55

BNP (pg/mL) e <10 0-100

pH 7,33 7,35-7,45

SO 2 (%) f 92,4 40-98

pO 2 (mmHg) g 71,5 80

pCO 2 (mmHg) h 42,6 35-48

cHCO 3 (Standard) (mmol/L) ı 21,3 22,5-26,9

a

Aspartate aminotransferase,

b

Alanine aminotransferase,

c

C-reactive protein,

d

Creatine kinase- MB,

e

Brain Natriuretic Peptide,

f

Oxygen saturation, g Partial oxygen pressure, h Partial carbondioxide pressure, ı Bicarbonate concentration

Signs and symptoms often include dyspnea, tachypnea,

bloody and frothy sputum, low oxygen saturation, pulmonary

crackles and signs of pulmonary edema on chest X-

ray. Differential diagnosis includes other causes of pulmonary

edema, such as aspiration of gastric content,

acute respiratory distress syndrome, congestive heart

failure, hypervolemia and pulmonary thromboembolism

(3). The absence of other etiologies listed above and the

onset of symptoms shortly after laryngospasm led to our

case being diagnosed with NPPE.

The treatment of NPPE comprises careful monitorization,

treatment of airway obstructions, and oxygen and mechanical

ventilatory support (1). There are different opinions

on the optimum medical treatment. While some

studies suggest that furosemide treatment is unnecessary,

since NPPE results from leaky capillaries and there is not

excessive fluid or hypervolemia (4). Others suggest furosemide

treatment due to its support of symptomatic and

radiological recovery (5). Even though there is no bronchospasm,

studies have shown that beta-agonist treatment

may heal pulmonary edema symptoms by making

removal of the fluid in the alveoli easier (6). Cases of

NPPE are usually recovered both clinical and radiological

quickly in 12-48 hours (5). Our case recovered quickly

with oxygen and non-invasive mechanical ventilator support,

diuretic, and inhaler beta-agonist treatment, and no

sequel was observed.

The prevention or early treatment of upper airway obstructions

would decrease the incidence of NPPE. Intraoperative

muscle relaxants, topical or spray lidocaine, or

steroids might be used (3). Moreover, further precautions

include careful aspiration of oropharyngeal secretions,



intubation of patients under very deep or very light anesthesia

when the risk of laryngospasm development is the

lowest and prophylactic continuous positive airway pressure

(CPAP) administration (7).

CONCLUSION

NPPE should be recalled quickly for patients who develop

respiratory failure following extubation, especially those

with a history of upper airway obstruction, as it can be life

threatening. Complete recovery is highly possible with

early diagnosis and treatment, and so all physicians

should be aware of this phenomenon.


None declared.


Concept - B.S., O.A.U.; Planning and Design - B.S.,

O.A.U.; Supervision - B.S., O.A.U.; Funding - O.A.U.;

Materials - B.S.; Data Collection and/or Processing - B.S.;

Analysis and/or Interpretation - B.S., O.A.U.; Literature

Review - B.S.; Writing - B.S., O.A.U.; Critical Review -

O.A.U.

YAZAR KATKILARI

Fikir - B.S., O.A.U.; Tasarım ve Dizayn - B.S., O.A.U.;

Denetleme - B.S., O.A.U.; Kaynaklar - O.A.U.; Malzemeler

- B.S.; Veri Toplama ve/veya İşleme - B.S.; Analiz

ve/veya Yorum - B.S., O.A.U.; Literatür Taraması - B.S.;

Yazıyı Yazan - B.S., O.A.U.; Eleştirel İnceleme - O.A.U.

REFERENCES

1. Liu R, Wang J, Zhao G, Su Z. Negative pressure pulmonary

edema after general anesthesia: A case report and

literature review. Medicine (Baltimore) 2019; 98:e15389.

[CrossRef]

2. Lorch DG, Sahn SA. Post-extubation pulmonary edema

following anesthesia induced by upper airway obstruction.

Are certain patients at increased risk? Chest 1986;

90:802-5. [CrossRef]

3. Pathak V, Rendon IS, Ciubotaru RL. Recurrent negative

pressure pulmonary edema. Clin Med Res 2011; 9:88-

91. [CrossRef]

4. Deepika K, Kenaan CA, Barrocas AM, Fonseca JJ, Bikazi

GB. Negative pressure pulmonary edema after acute upper

airway obstruction. J Clin Anesth 1997; 9:403-8.

[CrossRef]

5. Bhattacharya M, Kallet RH, Ware LB, Matthay MA. Negative-pressure

pulmonary edema. Chest 2016; 150:927-

33. [CrossRef]

6. Krodel DJ, Bittner EA, Abdulnour R, Brown R, Eikermann

M. Case scenario: acute postoperative negative pressure

pulmonary edema. Anesthesiology 2010; 113:200-7.

[CrossRef]

7. Galvis AG, Stool SE, Bluestone CD. Pulmonary edema

following relief of acute upper airway obstruction. Ann

Otol Rhinol Laryngol 1980; 89:124-8. [CrossRef]



OLGU SUNUMU

CASE REPORT

Diffuse Alveolar Hemorrhage Induced by

Sevoflurane

Sevofluran ile İndüklenen Diffüz Alveolar Hemoraji

Birsen Pınar Yıldız, Didem Görgün Hattatoğlu, Fulya Omak Kaya


Abstract

We present here a case of diffuse alveolar hemorrhage

(DAH) following exposure to inhaled anesthetic

sevoflurane. A 29-year-old male was admitted to the

plastic surgery department with gynecomastia. Surgery

was performed without complication under general

anesthesia with intravenously administered midazolam

(4mg), fentanyl and inhaled sevoflurane.

Immediate hypoxemia and massive hemorrhage was

detected at the end of the operation, and a chest

radiography revealed bilateral widespread alveolar

infiltrates. The serum hemoglobin level dropped by

2.5 g/dl (from 13 to 10.5 gr) in the postoperative

setting. The patient was treated with methylprednisolone

(1 gr) administered intravenously daily for 3 days.

The hypoxemia resolved and alveolar infiltrates on

the chest radiograph disappeared on the 4th day.

There is limited data in literature reporting on the

association between sevoflurane and DAH. None of

the predisposing factors or causative reasons for DAH

were detected in our case, and so it can be concluded

that the use of sevoflurane as an inhaled anesthetic

was responsible for the DAH.

Key words: Alveolar hemorrhage, sevoflurane, anesthesia.

Öz

İnhale anestezik sevoflurana maruz kaldıktan sonra

yaygın alveolar hemoraji (DAH) gelişen bir olguyu

sunmayı amaçladık. Burada sunulan hasta, jinekomasti

nedeniyle plastik cerrahiye başvuran 29 yaşında

bir erkek idi. Genel anestezi altında intravenöz midazolam

(4mg), fentanil ve inhale sevofluran ile komplikasyonsuz

cerrahi uygulandı. Ameliyat sonunda ani

hipoksemi ve masif kanama tespit edildi. Akciğer

grafisinde bilateral yaygın alveolar infiltratlar görüldü.

Ameliyat sonrası dönemde serum hemoglobin düzeyi

2,5 g/dl (13'ten 10,5 gr'a) düştü. Hastaya 3 gün

süreyle intravenöz olarak günlük metilprednizolon (1

gr) tedavisi verildi. Dördüncü günde hipoksemi düzeldi

ve akciğer grafisinde alveolar infiltratlar kayboldu.

Literatürdeki sınırlı veriler, sevofluran ve DAH

arasında bir ilişki olduğunu göstermektedir. Bizim

olgumuzda DAH' gelişimine neden olan sebepler ve

zemin hazırlayan faktörlerin hiçbiri saptanmadı. Bu

nedenle, DAH'dan inhale anestezik olarak sevofluran'ın

sorumlu olduğu düşünülmektedir.

Anahtar Sözcükler: Alveolar hemoraji, sevofloran,

anestezi.

Department of Pulmonology, University of Health Sciences,

Yedikule Chest Disease and Surgery Training and Research Hospital,

İstanbul, Türkiye

Sağlık bilimleri Üniversitesi, Yedikule Göğüs Hastalıkları ve

Göğüs Cerrahisi Eğitim ve Araştırma Hastanesi, Göğüs Hastalıkları

Anabilim Dalı, İstanbul


Correspondence (İletişim): Birsen Pınar Yıldız, Department of Pulmonology, University of Health Sciences, Yedikule Chest Disease

and Surgery Training and Research Hospital, İstanbul, Türkiye

e-mail: pinary70@yahoo.com

73


Diffuse alveolar hemorrhage (DAH) is a rare clinical syndrome

that presents in different clinics ranging from a

small amount of hemoptysis to life-threatening massive

hemoptysis. While connective tissue disorders such as

immune-mediated systemic vasculitis, Wegener's granulomatosis

and cytotoxic drugs may play a role in its etiology,

the exact pathogenesis is not yet known. (1) We

reported here a case of DAH attributed to the use of inhaled

anesthetic sevoflurane during a gynecomastia operation.

CASE

We present here the case of a 29-year-old male who was

admitted to the plastic surgery department due to gynecomastia.

His preoperative history, physical examination

and laboratory values were unremarkable. The patient

was New York Heart Association class I, and was monitored

with ECG, pulse oximetry (oxygen saturation) and

noninvasive blood pressure. Arterial blood pressure was

130/80 mmHg; Oxygen saturation was 98%, and his

heart and respiratory rate at baseline were 90 beats per

minute (bpm) and 15 breaths per minute (rpm), respectively.

Midazolam 1mg, lidocaine 1 mg/kg, thiopental

7mg/kg, rocuronium 0.6mg/kg and remifentanil 0.5

mg/kg were given to induce anesthesia and intubation

was performed. Anesthesia was maintained with 0 2 (%50),

air (%50) sevoflurane (%3) and remifentanil 0.125

mg/kg/min. Synchronous Intermittent Mandatory Ventilation

was maintained with tidal volume of 500 mL, a respiratory

rate of 12 rpm and an inspiration/expiration rate

of 1/2. Positive end-expiratory pressure (PEEP) was not

applied. Peak inspiratory pressure (P peak) was maintained

at 14–16cm H 20. During the intubation and postintubation,

the patient’s P peak and Plateau pressure did

not reach 30 cm H 20. After 40 minutes from the start of

anesthesia, the surgery was completed without complication.

Hemorrhage in the endotracheal tube was detected at the

end of the operation, when his vital signs were recorded

as blood pressure 110 /70 mmHg, temperature 36.4˚C,

heart rate 88/min and respiratory rate 32/min. The patient

was extubated after the hemorrhage was controlled.

Following extubation, the patient experienced a massive

hemorrhage and immediate hypoxemia, and was transferred

to the intensive care unit (ICU), where he was followed-up

extubated. Arterial blood gas tension was

measured at pH 7.39, PaCO 2 of 40.1 mm Hg and Pa0 2

49.5 mm Hg, with a saturation of 89%. Chest radiography

revealed bilateral widespread alveolar infiltrates,

and bilateral, centrally located ground-glass opacities

were noted on a computed tomographic angiography of

the chest, and pulmonary embolism was excluded (Figure

1). The serum hemoglobin level dropped by 2.5 g/dl

(from 13 to 10.5 gr) in the postoperative setting. The

platelet count was 154,000/µl, prothrombin time/ international

normalized ratio was 11.6 seconds/1.0, and

partial thromboplastin time was 24.8 seconds.

Serological testing was negative for HIV, viral hepatitis,

vasculitis and connective tissue diseases (Antinuclear

antibody, Anti glomerular basement membrane antibody,

Anti-JO1, Anti- DS DNA, Anti –SSA, Anti-SSB, Anti –

SCL70, Anti- SM/RNP, CCP, PANCA, cANCA), and a

urinalysis was normal. Daily methylprednisolone (1 gr)

treatment was administered intravenously for 3 days, and

the hypoxemia resolved and the alveolar infiltrates on

chest radiograph disappeared on the 4th day. Flexible

bronchoscopy has been evaluated and confirmed that

was no abnormality in the bronchial system. A bronchoalveolar

lavage cell count revealed polymorphonuclear

cells 2.9%; lymphocytes 1.1%; and alveolar macrophages

56%. The patient has no signs or symptoms related

to recurrence 4 months after recovery.

DISCUSSION

DAH has a wide clinical spectrum with the potential for

massive hemoptysis as well as asymptomatic radiologic

abnormalities. The majority of patients have a history of

hemoptysis, and decreased hemoglobin should serve as a

warning to physicians of the possibility of DAH. Patchy,

focal or diffuse alveolar filling processes can be seen on

chest radiography and CT. Bronchoscopy should be performed

to exclude other causes of hemoptysis and to

establish a clinical diagnosis of DAH. Progressive hemorrhagic

BAL found in serial samples is diagnostic for DAH.

(1) In our case, the serum hemoglobin level dropped by

2.5 g/dl. Clinical, bronchoscopic and radiographic evaluations

have been shown to confirm DAH and exclude

any other specific causative etiologies.

An inhalational anesthetic agent sevoflurane is used for

induction and maintenance of general anesthesia. Given

the immediate postoperative onset of alveolar hemorrhage

without reason, we can conclude that the causative

agent was inhaled sevoflurane. Alveolar hemorrhage

occurs in only a small number of cases, although sevoflorane

is associated with a number of respiratory side effects,

including cough, apnea, laryngeal spasm and respiratory

depression.


Diffuse Alveolar Hemorrhage Induced by Sevoflurane | Yıldız et al.

Lipid soluble volatile gases may increase alveolar permeability

by enhancing the arachidonic cascade in the cell

membrane, and increased oxidative stress and increased

inflammatory response (2,3), and previous studies have

suggested that the mechanism of sevoflurane causing

DAH may be related to this (4,5). Sevoflurane has also

been shown to inhibit platelet function and to reduce

platelet aggregation rates (6,7).

There is limited questionable data suggesting that

sevoflurane can induce alveolar hemorrhage (4,8,9), and

in the three previously-reported cases, the DAH was attributed

to the other causes. In the first case the patient

had a history of end-stage renal disease and cocaine use;

the patient had obstructive sleep apnea in the second

case, which can increase airway pressure (4,8); and the

third case was reported to show possible relationships

between sevoflurane and DAH with concurrent marijuana

use (9). The suggestion of this study is controversial, however,

as marijuana use has been previously reported as a

causative agent for DAH (9,10). In the above reports, the

association between drug use and DAH remains speculative.

Given the risk of airway obstruction in their patients,

Mersh et al. (11) and Hao et al. (12) both suggested that

alveolar hemorrhage may be due to negative pressure

pulmonary edema together with sevoflurane. In only two

cases were none of the predisposing factors or causative

reasons for DAH made available, as in our patient. Austin

et al. (5) reported on a young male who underwent urethral

stricture dilation via cystoscopy, while Cengiz et al.

(13) reported on a young male who underwent orthopedic

surgery. Interestingly, all of the above patients were

young 20–40-year-old males.

CONCLUSION

It can be concluded that the inhaled anesthetic drug

sevoflurane was the responsible agent that induced DAH

in our case, given the absence of any other disease or

administered agent. Although the mechanism of sevoflurane-causing

DAH cannot be fully clarified, it should be

kept in mind as the cause of life-threatening DAH.


None declared.


Concept - B.P.Y., D.G.H., F.O.K.; Planning and Design -

B.P.Y., D.G.H., F.O.K.; Supervision - B.P.Y., D.G.H.,

F.O.K.; Funding - B.P.Y., D.G.H.; Materials – F.O.K.,

B.P.Y.; Data Collection and/or Processing - F.O.K.; Analysis

and/or Interpretation - B.P.Y., D.G.H.; Literature

Review - D.G.H., B.P.Y.; Writing - D.G.H., B.P.Y.; Critical

Review - B.P.Y.

YAZAR KATKILARI

Fikir - B.P.Y., D.G.H., F.O.K.; Tasarım ve Dizayn - B.P.Y.,

D.G.H., F.O.K.; Denetleme - B.P.Y., D.G.H., F.O.K.;

Kaynaklar - B.P.Y., D.G.H.; Malzemeler - F.O.K., B.P.Y.;

Veri Toplama ve/veya İşleme - F.O.K.; Analiz ve/veya

Yorum - B.P.Y., D.G.H.; Literatür Taraması - D.G.H.,

B.P.Y.; Yazıyı Yazan - D.G.H., B.P.Y.; Eleştirel İnceleme -

H.D., F.D., B.P.Y.

REFERENCES

1. Lara AR, Schwarz MI. Diffuse alveolar hemorrhage. Chest

2010; 137:1164-71. [CrossRef]

2. Gentz BA, Malan TP Jr. Renal toxicity with sevoflurane: a

storm in a teacup? Drugs 2001; 61:2155-62. [CrossRef]

3. Shayevitz JR, Traystman RJ, Adkinson NF, Sciuto AM,

Gurtner GH. Inhalation anesthetics augment oxidantinduced

pulmonary vasoconstriction: evidence for a

membrane effect. Anesthesiology 1985; 63:624-32.

[CrossRef]

4. Kim JP, Park JJ, Kim NJ, Woo SH. A case of diffuse alveolar

hemorrhage after tonsillectomy -A case report-. Korean

J Anesthesiol 2012; 63:165-8. [CrossRef]

Figure 1: Chest x-ray and CT showing bilateral infiltrates at initial presentation

(a, c), Chest x-ray and CT 3 days after pulse steroid (b, d)

5. Austin A, Modi A, Judson MA, Chopra A. Sevoflurane

induced diffuse alveolar hemorrhage in a young patient.

Respir Med Case Rep 2016; 20:14-5. [CrossRef]

6. Hirakata H, Ushikubi F, Narumiya S, Hatano Y, Nakamura

K, Mori K. The effect of inhaled anesthetics on the

platelet aggregation and the ligand-binding affinity of the



platelet thromboxane A2 receptor. Anesth Analg 1995;


7. Doğan IV, Ovali E, Eti Z, Yayci A, Göğüş FY. The in vitro

effects of isoflurane, sevoflurane, and propofol on platelet

aggregation. Anesth Analg 1999; 88:432-6.

[CrossRef]

8. Khanna AK, Cummings KC 3rd. Pulmonary hemorrhage

in an outpatient ophthalmic anesthesia setting - it's never

"just a cataract". J Anaesthesiol Clin Pharmacol 2012;


9. Kim CA, Liu R, Hsia DW. Diffuse alveolar hemorrhage

induced by sevoflurane. Ann Am Thorac Soc 2014;


10. Murray AW, Smith JD, Ibinson JW. Diffuse alveolar hemorrhage,

anesthesia, and cannabis. Ann Am Thorac Soc

201; 11:1338-9. [CrossRef]

11. Mersh R, Ross C. Postoperative diffuse alveolar haemorrhage:

insidious negative pressure or sevoflurane induced?

BMJ Case Rep 2018; 2018:bcr2017222010. [CrossRef]

12. Hao D, Basnet S, Melnick S, Kim J. Negative pressure

pulmonary edema-related diffuse alveolar hemorrhage

associated with Sevoflurane and cigarette smoking. J

Community Hosp Intern Med Perspect 2019; 9:247-51.

[CrossRef]

13. Cengiz O, Kivrak A, Yegen M, Demir M. Sevoflurane induced

diffuse alveolar haemorrhage in a young patient

after orthopedic surgery: A case report. Niger J Clin Pract

2020; 23:120-2. [CrossRef]



OLGU SUNUMU

CASE REPORT

A Case Report of Tuberculous Lymphadenitis

and Tuberculous Pleural Effusion

Accompanied by Splenic Tuberculosis

Splenik Tüberkülozun Eşlik Ettiği Tüberküloz Lenfadenit ve Tüberküloz Plörezi:

Bir Olgu Sunumu

Gamze Kayasuyu 1 , Ceyda Anar 1 , Süheyla Uygur 1 , Betül Doğan 1 , Muzaffer Onur Turan 1 ,

Bunyamin Sertogullarindan 1 , Ebru Cakir 2


Abstract

Spleen tuberculosis has occasionally been described

in literature, mostly in immunocompromised patients

with various risk factors. The presence of spleen involvement

and splenomegaly makes this type of Mycobacterium

tuberculosis infectiondifficult to diagnose.

A 56-year-old woman who had mediastinal and

abdominal lymphadenopathies with splenomegaly

and was investigated with a pre-diagnosis of lymphoma,

and with no significant medical history, presented

with complaints of fever and malaise. Here,

we present a case of tuberculous lymphadenitis and

pleuritis accompanied by splenic tuberculosis with the

appearance of granulomatous inflammation on pleural

and spleen biopsy. Tuberculosis should be kept in

mind as a differential diagnosis in diseases accompanied

by a fever of unknown origin and lymphadenopathy.

Key words: Tuberculosis, lymphadenit, spleen, pleural

effusion.

1 Department of Pulmonology, Iżmir Katip Çelebi Üniversity Atatürk

Training and Research Hospital, İzmir, Türkiye

2 Department of Patology, Iżmir Katip Çelebi Üniversity Atatürk

Training and Research Hospital, İzmir, Türkiye

Öz

Dalak tüberkülozu, literatürde zaman zaman, çoğunlukla

çeşitli risk faktörleri olan bağışıklığı baskılanmış

bireylerde tanımlanmıştır. Dalak tutulumu ve splenomegalinin

olması, bu tür Mycobacterium tuberculosis

enfeksiyonunun teşhis edilmesini zorlaştırır. Splenomegali

ile birlikte mediastinal ve abdominal lenfadenopatileri

olan ve lenfoma ön tanısı ile araştırılan,

önemli bir tıbbi öyküsü olmayan 56 yaşında kadın

hasta ateş, halsizlik yakınması ile başvurdu. Alınan

plevral ve dalak biyopsisinde granülomatöz inflamasyonun

ortaya çıktı. Burada, dalak tüberkülozunun

eşlik ettiği tüberküloz lenfadenit ve plörit olgusunu

sunuyoruz. Tüberküloz, nedeni bilinmeyen ateş ve

lenfadenopatilerin eşlik ettiği hastalıklarda ayırıcı tanı

olarak hala akılda tutulmalıdır.

Anahtar Sözcükler: Tüberküloz, lenfadenit, dalak,

plevral effüzyon.

1 İzmir Katip Çelebi Üniversitesi Atatürk Eğitim ve Araştırma

Hastanesi, Göğüs Hastalıkları Bölümü, İzmir

2 İzmir Katip Çelebi Üniversitesi Atatürk Eğitim ve Araştırma

Hastanesi, Patoloji Bölümü, İzmir


Correspondence (İletişim): Ceyda Anar, Department of Pulmonology, İzmir Katip Çelebi Üniversity Atatürk Training and Research

Hospital, İzmir, Türkiye

e-mail: drceydaanar@hotmail.com

77


Despite the medical advances in the diagnosis and treatment

of infectious diseases, tuberculosis is still a health

issue of concern in developing countries. In countries

where cases of tuberculosis are common, all forms of the

disease may frequently be detected, while in developed

countries extrapulmonary cases represent a very small

proportion of the total. Although the spleen is the third

most affected organ in miliary or disseminated tuberculosis

after the lungs and liver, isolated splenic tuberculosis is

a rare manifestation of the disease (1). Symptoms of

splenic tuberculosis are often nonspecific and misleading,

and even asymptomatic cases can often complain of

fever, stomachache and weight loss (2). The early diagnosis

of splenic tuberculosis is not possible due to the

unclear symptomatology of the disease, while Mycobacterium

tuberculosis infections can be difficult to diagnose

due to bacterial sequestrations in the spleen. Carcinoma,

metastatic tumor, lymphoma, hemangioma and abscess

of the spleen are among the differential diagnoses, and

false diagnoses are also likely in high rates if there is no

history of tuberculosis of other organs.

We present here the case of a 56-year-old woman with

no significant medical history who had mediastinal and

abdominal lymphadenopathies together with splenomegaly,

and who had been investigated with a preliminary

diagnosis of lymphoma. We present a case of pleural

effusion with pleural and splenic biopsy results of granulomatous

inflammation leading to splenic tuberculosis,

with accompanying tuberculous lymphadenitis and pleurisy.

Tuberculosis must be kept in mind as a differential

diagnosis when presented with fever of unknown origin

and diseases accompanied by lymphadenopathies.

CASE

A 56-year-old woman presented with a 4-month history

of excessive sweating and weight loss. A thoracicabdominal

computed tomography (CT) revealed many

lymph nodes with a short axis of 3 cm in the mediastinum

and abdominal lymphadenopathies, together with splenomegaly.

Considering the preliminary diagnosis of lymphoma,

an inguinal lymph node excisional biopsy was

carried out (Figure 1a and b), and the histopathological

result of the biopsy revealed noncaseating granulomatous

lymphadenitis. The patient was thus referred to our chest

diseases unit with preliminary diagnoses of sarcoidosis

and tuberculosis. A Mantoux test/tuberculin skin test (PPD

skin test) was performed on the patient, whose histopathological

diagnosis was granulomatous lymphadenitis,

for the differential diagnosis of tuberculosis and sarcoidosis.

A tuberculin skin test showed no induration, and

EBUS was planned for culture material from the mediastinal

lymph nodes. The patient did not attend the clinic at

this time due to the pandemic, and applied to the chest

diseases clinic due to shortness of breath approximately 2

months later. Pleural effusion had formed in the left lung

approximately from the 2nd anterior rib on the posterior

in an anterior Chest x-ray (Figure 2). The patient was

hospitalized and a physical examination revealed a moderate

and weak general condition. Multiple lymphadenopathies

were detected bilaterally in the cervical, inguinal

and axillary areas that were mobile, firm and painless,

and with no signs of inflammation or induration on the

skin. Tenderness and Traube’s Space were evaluated as

closed. Laboratory examinations revealed a hemoglobin

level of 10.6 g/dl, hematocrit of 34%, leukocytes of

6590/mm 3 and platelets of 336000/mm 3 . The erythrocyte

sedimentation rate was determined as 12 mm·h−1.

The biochemical parameters values were BUN of 13

mg/dl, Creatinine of 0.6 mg/dl, Na of 137 meq/L, K of

5.0 meq/L, Ca of 7.5 mg/L, Albumin of 2 g/dl, AST of

26 IU/L, ALT of 13 IU/ L and LDH of 278 IU/L. The patient

underwent thoracentesis, and the pleural fluid was

an exudative effusion, the result of an acid-fast bacilli

(ARB) test of which was negative. The adenosine deaminase

(ADA) levels of the pleural effusion were 22 IU/L. A

closed needle biopsy was performed on the patient.

While there were many granuloma structures in lymphoplasmacytic

inflammatory cell infiltrations, there was no

observation of necrosis in the granuloma structures in the

pathology results of the pleural biopsy. Schaumann bodies

were observed in some of the giant cells, the presence

of which indicated possible sarcoidosis as a clinical prediagnosis.

Since these findings were not entity-specific in

terms of causes of granulomatous inflammation (Figure

3), the case was evaluated from a clinical, microbiological,

and serological point of view. Further examinations

revealed noticeable symptoms of dyspnea in the foreground,

and an evacuating thoracentesis was performed.

Due to repetitive pleural fluid collections, a pleura can

was attached. While lymphoma was ruled out by hematology,

PET/CT revealed multiple low-to-moderate hypermetabolic

lymph nodes with conglomeration and

chain-like extension in the cervical-thoracic region, abdomen

and pelvis. The spleen was also significantly increased

in size, and in addition to a significant diffuse F-

18 FDG uptake in the parenchyma of the spleen, a

pathological F-18 FDG uptake compatible with the capsular

millimetric hypodense area was detected at the level


A Case Report of Tuberculous Lymphadenitis and Tuberculous Pleural Effusion Accompanied by Splenic Tuberculosis | Anar et al.

of the inferior end of Segment 6 in the liver (Figure 4a

and b). A spleen biopsy was performed on the patient,

and a biopsy sample taken from the cervical lymph node

was sent to the microbiology department for tuberculosis

culture. There was no ARB identified in a direct examination

of the lymph node. While waiting for the culture result,

caseifying granulomas were observed in the spleen

biopsy (Figure 5). The patient was started on antituberculous

treatment with a diagnosis of splenic tuberculosis,

tuberculous lymphadenitis and pleural tuberculosis. No

growth was detected in the lymph node tuberculosis culture

of the patient who was now in the 2nd month of

treatment.

DISCUSSION

Tuberculosis is a multisystemic disease, the most common

form of which is pulmonary tuberculosis. Extrapulmonary

disease accounts for approximately 15–20% of all cases

of tuberculosis (3). Splenic tuberculosis was first described

in literature by Coley in 1846, but this unusual form is

rarely reported as the primary involvement in literature.

HIV-infected or immunocompromised patients are reported

to be at high risk of splenic tuberculosis, and HIV

infection has been found to underlie many reported cases

of splenic tuberculosis abscesses (4). For this reason,

spleen involvement was thought to occur only in immunocompromised

individuals, although there have been a

few case reports of splenic tuberculosis in immunocompetent

patients (5). Sharma et al. (6) and Gupta et al. (7)

reported rare cases of splenic abscess in an immunocompromised

and immunocompetent patient. The case

presented here is extremely rare. The patient had no history

of tuberculosis, and no immunosuppressive conditions

have been shown to cause such infections. The

splenic involvement of tuberculosis is more common in

men, and usually in the 19–53 years age group (8). In

most cases, a fever of unknown origin is the typical

presentation (9). The most common symptoms presented

by patients are fever (82.3%), fatigue, weight loss

(44.12%), and splenomegaly (13.2–100%) (10). There

are no specific symptoms for a diagnosis of splenic tuberculosis.

Tuberculosis of the spleen is very rare and difficult

to diagnose in an immunocompetent individual. The

main complaints in our case – a 56-year-old female –

were fever, malaise, anorexia, abdominal pain and

shortness of breath. Most cases present as a splenic abscess,

but the presence of ascites (11) together with a

splenic mass (12) has also been reported. Splenic TB may

be associated with the presence of abdominal and mediastinal

lymphadenopathy, psoas abscess and vertebral

tuberculosis (13). In our case, pleural effusion accompanied

the splenic tuberculosis, along with abdominal and

mediastinal lymphadenopathies. Isolated cases of splenic

tuberculosis have been reported in the literature (14-16).

Despite their reliability, common methods such as Ultrasound

and CT offer only limited success in distinguishing

lesions from primary or metastatic tumors of the spleen.

The rate of misdiagnosis is high if there is no history of

tuberculosis in other organs. Although the presence of

splenomegaly with abdominal lymph nodes in our case

initially suggested lymphoma as a diagnosis, the granulomatous

result of the biopsy taken from both the lymph

node and spleen ruled this condition out. Although imaging

methods are helpful in the diagnosis of tuberculosis,

whether pulmonary or extrapulmonary, a definitive diagnosis

is made histopathologically and/or microbiologically.

Abdominal ultrasound is the first imaging modality of

choice, as it is cost-effective and can aid in the characterization

of the lesion. Lesions of the spleen typically present

as multiple hypoechoic lesions that may be tuberculomas

or abscesses. CT is the next best modality for the

identification of lesions of the spleen, and also for the

detection of concurrent lesions in the chest and other

parts of the abdomen. Many patterns such as nodular,

pseudotumor and oval appearances, have been identified

in splenic tuberculosis on CT scans, and five types of

splenic tuberculosis can be identified based on pathomorphological

manifestations, including miliary tuberculosis,

nodular tuberculosis, tuberculous splenic abscess,

calcific tuberculosis and mixed type tuberculosis (17).

Diagnosis is confirmed by a histopathological examination

of the lesions. Fine-needle aspiration biopsy, laparoscopic

or imaging-guided needle biopsy (CNB), or splenectomy

specimens have been examined for diagnostic

confirmation in different reports (18). In the abdominal

USI performed in our case, the size of the spleen was

measured at approximately 28 mm, and the parenchyma

structure was observed as normal. There were, however,

lymph nodes present in the abdomen. The patient, who

was thought to have mostly lymphoma based on her

splenomegaly, was found to have splenomegaly with

abdominal lymphopathies in her abdominal CT. Imaging

methods cannot contribute to a direct diagnosis, and so a

histopathological diagnosis is required. In our case, an

FDG uptake was also present in the spleen and mediastinal,

and abdominal lymphadenopathies on PET-CT,

however, these do not indicate a specific disease, and

can only guide clinicians to the appropriate biopsy site.



Since lymphoma was considered as a preliminary diagnosis,

no culture was sent from the biopsies, and the presence

of splenomegaly made it difficult for us to come to a

diagnosis. A sample, however, was sent from the cervical

lymph node for culture. As with pulmonary tuberculosis,

antituberculous therapy is the primary treatment approach

to spleen tuberculosis. Treatment regimens, which typically

last six to nine months, are considered satisfactory, and

a splenectomy can be applied to patients who do not

respond to medical treatment or in the presence of an

abscess in the spleen (17).

Figure 1a and b: Many lymph nodes with a short axis of 3 cm in the

mediastinum

Figure 3: Granulomatous inflammation on pleural biopsy (H&E, X100)

Figure 2: Pleural effusion on the left lung

One study in literature reported a case of isolated spleen

TB in which a fever of unknown origin was examined, and

was not detected in any other morphological image examination

other than FDG-PET (19). In our case, biopsy

samples taken from the lymph nodes, pleura and spleen

were reported as granulomatous. Histopathologically,

caseification was also observed in the spleen biopsy.

Figure 4a and b: PET/CT revealed multiple low-to-moderate hypermetabolic

lymph nodes with conglomeration and chain-like extension in

the cervical, thoracic region, abdomen and pelvis, and a significantly

increased spleen size was detected in addition to significant diffuse F-18

FDG uptake in the parenchyma of the spleen


A Case Report of Tuberculous Lymphadenitis and Tuberculous Pleural Effusion Accompanied by Splenic Tuberculosis | Anar et al.

REFERENCES

1. Nayyar V, Ramakrishna B, Mathew G, Williams RR,

Khanduri P. Response to antituberculous chemotherapy

after splenectomy. J Intern Med 1993; 233:81-3.

[CrossRef]

2. Udgaonkar U, Kulkarni S, Shah S, Bhave S. Asymptomatic,

isolated tubercular splenic abscess, in an immunocompetent

person. Indian J Med Microbiol 2010;


Figure 5: Caseative necrotizing granulomatous inflammation of the

spleen parenchyma (H&E, X100)

In conclusion, although fever of unknown origin, weight

loss, night sweats, and multiple lymphadenomegaly and

lymphoma are considered in the differential diagnosis of

splenomegaly, it should be kept in mind that there are

unusual manifestations of tuberculosis that can mimic any

disease, and that may create difficulties in the differential

diagnosis. It is necessary to emphasize here that if clinical

and radiological findings suggest tuberculosis, it will be

useful to perform microbiological examinations to determine

the causative agent from materials taken from the

lymph node, pleura or any organ other than the lung.


None declared.


Concept - G.K., C.A., S.U., B.D., M.O.T., B.S., E.C.;

Planning and Design - G.K., C.A., S.U., B.D., M.O.T.,

B.S., E.C.; Supervision - G.K., C.A., S.U., B.D., M.O.T.,

B.S., E.C.; Funding - G.K., C.A., B.D., M.O.T.; Materials

- G.K., C.A., S.U.; Data Collection and/or Processing -

G.K., C.A., S.U., B.D., E.C.; Analysis and/or Interpretation

- C.A., S.U., B.S., E.C.; Literature Review - C.A., S.U.,

B.S., E.C.; Writing - C.A., S.U., B.S., E.C., B.D.; Critical

Review - C.A., S.U., G.K., E.C.

YAZAR KATKILARI

Fikir - G.K., C.A., S.U., B.D., M.O.T., B.S., E.C.; Tasarım

ve Dizayn - G.K., C.A., S.U., B.D., M.O.T., B.S., E.C.;

Denetleme - G.K., C.A., S.U., B.D., M.O.T., B.S., E.C.;

Kaynaklar - G.K., C.A., B.D., M.O.T.; Malzemeler - G.K.,

C.A., S.U.; Veri Toplama ve/veya İşleme - G.K., C.A.,

S.U., B.D., E.C.; Analiz ve/veya Yorum - C.A., S.U., B.S.,

E.C.; Literatür Taraması - C.A., S.U., B.S., E.C.; Yazıyı

Yazan - C.A., S.U., B.S., E.C., B.D.; Eleştirel İnceleme -

C.A., S.U., G.K., E.C.

3. Hamizah R, Rohana AG, Anwar SA, Ong TZ, Hamazaini

AH, Zulkarnaen AN. Splenic tuberculosis presenting as

pyrexia of unknown origin. Med J Malaysia 2007;

62:70-1.

4. Pramesh CS, Tamhankar AP, Rege SA, Shah SR. Splenic

tuberculosis and HIV-1 infection. Lancet 2002; 359:353.

[CrossRef]

5. Chandra S, Srivastava DN, Gandhi D. Splenic tuberculosis:

an unusual sonographic presentation. Int J Clin Pract

1999; 53:318-9.

6. Sharma S, Dey AB, Agarwal N, Nagarkar KM, Gujral S.

Tuberculosis: a rare cause of splenic abscess. J Assoc

Physicians India 1999; 47:740-1.

7. Gupta A, Hunjan PS, Jain SK, Kaza RC, Kumar V. Tubercular

splenic abscess in an immunocompetent patient: a

rare entity. Southeast Asian J Trop Med Public Health

2006; 37:1196-8.

8. Pottakkat B, Kumar A, Rastogi A, Krishnani N, Kapoor VK,

Saxena R. Tuberculosis of the spleen as a cause of fever

of unknown origin and splenomegaly. Gut Liver 2010;

4:94-7. [CrossRef]

9. Ho PL, Chim CS, Yuen KY. Isolated splenic tuberculosis

presenting with pyrexia of unknown origin. Scand J Infect

Dis 2000; 32:700-1. [CrossRef]

10. Rhazal F, Lahlou MK, Benamer S, Daghri JM, Essadel E,

Mohammadine E, et al. Splenomegaly and splenic

pseudotumor due to tuberculosis: six new cases. Ann

Chir 2004; 129:410-4. [CrossRef]

11. Lim J, Yu JS, Hong SW, Chung JJ, Kim JH, Kim KW. A

case of mass-forming splenic tuberculosis: MRI findings

with emphasis of diffusion-weighted imaging characteristics.

J Korean Med Sci 2011; 26:457-60. [CrossRef]

12. Lonkar Y, Parikh S, Kumar S, Diwan S, Bhake A. Splenic

tuberculosis presenting as ascites in immunocompetant

patient. Ann Med Health Sci Res 2013; 3:116-8.

[CrossRef]

13. Sharma SK, Smith-Rohrberg D, Tahir M, Mohan A, Seith

A. Radiological manifestations of splenic tuberculosis: a

23-patient case series from India. Indian J Med Res 2007;

125:669-78.



14. Wangai F, Achieng L, Otieno G, Njoroge J, Wambaire T,

Rajab J. Isolated splenic tuberculosis with subsequent

paradoxical deterioration: a case report. BMC Res Notes

2017; 10:162. [CrossRef]

15. Gupta P, Dhaka N, Rohilla M. Isolated splenic tuberculosis

presenting as an unusual splenic mass . Int J Mycobacteriol

2018; 7:397-8. [CrossRef]

16. Dhibar DP, Chhabria BA, Gupta N, Varma SC. Isolated

splenic cold abscesses in an immunocompetent individual.

Oman Med J 2018; 33:352-5. [CrossRef]

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al. Isolated splenic tuberculosis: a case report. World J

Gastrointest Pathophysiol 2010; 1: 109-11. [CrossRef]

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a case report. Int J Infect Dis 2009; 13:e273-5.

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Isolated splenic tuberculosis detected only on FDG-PET.

BJR Case Rep 2017; 3:20150238. [CrossRef]



OLGU SUNUMU

CASE REPORT

Secondary Brain Abscess in a Patient

Followed Up with Lung Abscess

Akciğer Apsesi ile Takip Edilen Hastada İkincil Beyin Apsesi

Özlem Ataoğlu 1 , Pinar Yildiz Gulhan 2 , Mehmet Fatih Elverisli 3 , Ege Güleç Balbay 2


Abstract

Respiratory tract infections are common in Down

syndrome. A 24-year-old male patient with Down

syndrome with concurrent diabetes mellitus underwent

antibiotic treatment with a diagnosis of lung

abscess in an external center with complaints of fever

and vomiting, but was referred to us after his symptoms

did not regress. Despite the improvement in the

lung abscess noted in a radiological examination, the

patient was identified with a brain abscess upon an

examination due to the continuation of fever, vomiting

and the onset of headache. Particular attention

should be paid to additional abscess foci such as

brain abscess in cases with lung abscess with an

underlying comorbidity.

Key words: Down syndrome, lung abscess, brain

abscess.

1 Düzce Ataturk State Hospital, Düzce, Türkiye

2 Department of Chest Diseases, Düzce University Faculty of

Medicine, Düzce, Türkiye

3 Ünye State Hospital, Ordu, Türkiye

Öz

Down sendromunda solunum yolu enfeksiyonları sık

olarak görülmektedir. Diabetes mellitusu da olan

Down sendromlu 24 yaşındaki erkek hasta, ateş ve

kusma şikayetleri ile gittiği dış merkezde akciğer apsesi

tanısıyla antibiyotik tedavisi almış, ancak semptomlarının

gerilememesi üzerine tarafımıza sevk edilmiştir.

Radyolojik incelemede akciğer apsesinde düzelme

olmasına rağmen ateş, kusmanın devam etmesi

ve baş ağrısının da başlaması üzerine yapılan incelemelerde

hastanın beyin apsesinin de olduğu saptandı.

Özellikle altta yatan ek hastalığı olan akciğer

apseli olgularda beyin apsesi gibi ek apse odakları

açısından dikkatli olunmalıdır.

Anahtar Sözcükler: Down sendromu, akciğer apsesi,

beyin apsesi.

1 Düzce Atatürk Devlet Hastanesi, Düzce

2 Düzce Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim

Dalı, Düzce

3 Ünye Devlet Hastanesi, Ordu


Correspondence (İletişim): Özlem Ataoğlu, Düzce Ataturk State Hospital, Düzce, Türkiye

e-mail: dr.ozlemozturk@outlook.com

83


Down Syndrome (DS) is the most common of the chromosomal

abnormalities, with an incidence of approximately

1/800 (1). The relationship between DS and 21st chromosome

abnormalities was first identified in 1959 (2).

Craniofacial anomalies, hypotonia, cognitive disorders,

Alzheimer's dementia (3), gastrointestinal malformations,

congenital heart defects, respiratory tract diseases, autoimmunity,

thyroid dysfunction and hematological disorders

can all be seen in DS (4)

Respiratory diseases are the most common cause of

death in DS patients of any age (5), with infection and

acute respiratory distress syndrome being the most common

respiratory tract diseases (6). The respiratory tract in

DS patients is usually narrow due to hypoplasia, macroglossia,

narrowing of the nasopharynx, enlarged tonsils,

choanal stenosis, shortening of the palate, subglottic

stenosis, laryngomalacia, tracheomalacia, gastroesophageal

reflux, obesity, and congenital anomalies in the

bronchi and airways (7), leading to recurrent respiratory

tract infections (8). Respiratory tract diseases can also be

linked to delayed motor function, structural anomalies of

the oronasal passages, and disorders in innate and acquired

immunity (9,10). Congenital anomalies of the

respiratory tract, delayed motor function and structural

anomalies of the oral-nasal passages contribute not only

to infection, but also to chronic aspiration (9).

These problems in DS patients may also contribute to the

formation of lung abscesses, defined as a limited area of

infectious parenchymal necrosis that forms in one or

more suppurative spaces (10).

More than 90% of cases present with polymicrobial bacteria

(11). Among the isolates that appear dominant in

lung abscess, Bacteroides fragilis, Fusobacterium capsulatum

and necrophorum, anaerobic peptostreptococcus

and microearophillic streptococcus are anaerobes. While

the aerobics are Staphylococcus aureus, Streptococcus

pyogenes and pneumonia, Klebsiella pneumonia, Pseudomonas

aeruginosa, Hemophilus influenza type-B, Acinetobacter

spp, Escherichia coli and legionella (12). Identifying

differences in clinical manifestations and in microbiological

isolates of lung abscesses may have important

implications in guiding clinical diagnostic evaluations and

empirical antimicrobial management, particularly in immunocompromised

hosts (13)

Clinically, lung abscess may develop silently within a few

weeks, and most commonly tachypnea, cough and fever.

They can be identified on a posteroanterior chest (PA)

radiography, while ultrasound and computed tomography

(CT) can confirm the diagnosis. Up to 90% can be treated

with intravenous (IV) antibiotics (10).

Brain abscesses may occur secondary to neurosurgical

procedures, or primarily due to the hematogenous spread

of sinusitis, periodontal infection, mastoiditis and pulmonary

infections (14). Periodontal disease is an independent

risk factor for brain abscess (15). When compared to

typically developed people and those with intellectual

disabilities, the incidence of early progressive periodontal

disease is increased in patients with DS (16).

In cases with lung abscess due to an underlying disease,

the risk of brain abscesses may be increased. We present

here a case of DS with abscesses in both the brain and

lungs.

CASE

A 24-year-old male patient with a known diagnosis of DS

and diabetes mellitus (DM) was diagnosed with lung

abscess at an external center with complaints of fever and

nausea, and was treated with IV sulbactam-ampicillin for

4 days and piperacillin-tazobactam (PTZ) for 8 days. He

was referred to us after his fever and nausea continued,

and complained of headache as well as fever and nausea

upon admission. A PA chest X-ray of the patient revealed

an appearance consistent with a cavity above the diaphragm

of the right lung (Figure 1a). A thorax CT of the

right lower lobe laterobasal segment revealed a consolidated

area of 3.2x1.2 cm and a cavitary appearance that

was thought to open into a bronchus of 1x0.5 cm in size,

suggesting an abscess.

The patient was consulted to department of infectious

diseases in terms of arranging his antibiotic therapy at his

hospitalization. The patient, who was in a good general

condition, underwent IV antibiotic therapy for a total of

12 days in another center, was recommended to take

cultures and to continue PTZ antibiotic therapy. Bronchoscopy

was performed on the patient, who could not

produce sputum due to continued fever. There was no

reproduction in the culture of a bronchial lavage taken.

Regression was observed in the control PA chest radiograph

(Figure 1b). No control thorax CT was taken, as

the lesion was noted to have improved in the chest X-ray

taken after the treatment. As the patient’s fever continued

on the 16th day of PTZ treatment, the patient was started

on imipenem. Neurology consultation was requested for

the patient, whose general condition was good but whose

complaints of headache and vomiting continued. Cranial

magnetic resonance imaging (MRI) of the patient revealed

a space-occupying lesion at the level of the parie-


Secondary Brain Abscess in a Patient Followed Up with Lung Abscess | Ataoğlu et al.

tooccipital junction, and a thin, cerebral abscess with

regular contrast enhancement on the periphery (Figure 2).

Since imipenem lowered the seizure threshold, it was

substituted by meropenem. A further neurosurgery consultation

was requested, and it was decided to operate on

the brain abscess. A biopsy taken at the end of the operation

revealed cerebrum tissues showing liquefaction necrosis.

After the surgery, the patient's complaints of fever,

nausea, vomiting and headache subsided.

DISCUSSION

Lung abscesses are necrotic cavitary lesions of the lung

parenchyma and are usually caused by anaerobic bacteria

or mixed flora, occurring typically after aspiration.

Primary lung abscesses occur in healthy individuals without

underlying disease and are usually solitary, while

secondary lung abscesses occur in patients with underlying

or predisposing conditions, and may be multiple. The

first finding is usually the cavity view, which gives the airfluid

level on the PA chest x-ray. Typically, the cavity wall

is thick and irregular, and there is usually a surrounding

pulmonary infiltrate that requires prolonged antibiotic

therapy (17). In our case, the diagnosis was made by PA

Chest X-ray.

In the presence of lung abscesses, it is difficult to isolate

anaerobic bacteria under these conditions, as most respiratory

tract samples (sputum or bronchoscopy aspirates)

are contaminated with upper airway flora, and are therefore

unsuitable for anaerobic culture (12). In our case,

concurring with literature, there was no reproduction in

the culture in the lavage sample taken with bronchoscopy.

Congenital anomalies of the respiratory tract, delayed

motor function, structural anomalies of the oronasal passages,

and defects in innate and acquired immunity may

have contributed to the predisposition of our DS patient

to respiratory tract infections, chronic aspiration and the

formation of lung abscess. A review of literature revealed

no other case of lung abscess in DS.

Figure 1a and b: First chest X-ray of the patient at hospital admission (a),

Control chest x-ray of the patient (b)

Figure 2: Parietooccipital region abscess appearance



Brain abscesses are necrotic focal areas with a membrane

surrounding the brain parenchyma. They are usually

caused by bacteria or fungi, and rarely as a result of

trauma, and have high morbidity and mortality rates.

Brain abscesses may occur secondary to neurosurgical

procedures, or primarily due to hematogenous spread

such as through pulmonary infections (14). In our case,

the abscess likely spread from the lung abscess via a

hematogenous route. In our case, the compatibility of the

pathological outcome with liquefaction necrosis, the bacterial

or fungal infection, and the response to antibiotic

treatment suggest that the infection was of bacterial origin.

In about two-thirds of cases, symptoms last 2 weeks or

fewer. Diagnoses are made on average 8 days after the

onset of symptoms. Most of the symptoms of a brain

abscesses are nonspecific, leading to a delay in diagnosis,

and most of the symptoms linked to space-occupying

lesions are related to its size and location. The triad of

fever, headache and focal neurologic deficit is seen in

less than half of the patients. The most common symptoms

are headache, mental status changes, focal neurological

deficits, fever, seizures, nausea and vomiting (18).

Our case had complaints of fever, headache and nausea.

Periodontal disease is often associated with DM, and may

remain clinically silent. Periodontal disease is an independent

risk factor for brain abscess (15). Our patient

also had both DS and DM disease, both of which are

linked to periodontal disease, which also featured in our

patient’s history.

MRI is the imaging modality of choice for diagnosis. In

the early period, it is sensitive in revealing lesions in the

brain, and especially in the brain stem, and also any

necrosis of the lesion. It provides greater contrast between

cerebral edema and the brain, and it is more sensitive in

the detection of the spread of inflammation to the ventricles

and subarachnoid space (19). In our case, the brain

abscess was observed in a brain MRI taken together at

the time of a neurological evaluation due to prolonged

nausea and vomiting. Treatment is usually both medical

and surgical (14), and our case was both operated and

placed on long-term antibiotic therapy.

Radoi et al. (20), in a retrospective study involving 52

consecutive brain abscess patients, the authors reported

the most common cause of brain abscess to be hematogenous

spread. In our case, we concluded that the brain

abscess developed secondary to hematogenous spread

from the lung abscess.

Our brain abscess case had both DS and DM, which led

to a disorder in the immune system, and these two diseases

increase the risk of periodontal disease. Since brain

abscesses are mostly spread secondarily via the hematogenous

route, we concluded that in our case the brain

abscess may have spread from the lung abscess via the

hematogenous route. A review of literature revealed no

cases of brain abscess secondary to lung abscess in DS,

or brain abscesses without lung abscesses.

It should be kept in mind that a brain abscess may also

be present in cases stated on antibiotic therapy for a lung

abscess and that demonstrate radiological improvement,

but with continued fever, vomiting and headache. It

should be further noted that in cases with lung abscess

with an underlying comorbidity, a brain abscess or other

abscess foci may also be present.


None declared.


Concept - Ö.A., P.Y.G., M.F.E., E.G.B.; Planning and

Design - Ö.A., P.Y.G., M.F.E., E.G.B.; Supervision - Ö.A.,

P.Y.G., M.F.E., E.G.B.; Funding - Ö.A.; Materials - Ö.A.;

Data Collection and/or Processing - Ö.A.; Analysis

and/or Interpretation - Ö.A., P.Y.G.; Literature Review -

Ö.A., P.Y.G.; Writing - Ö.A.; Critical Review - Ö.A.

YAZAR KATKILARI

Fikir - Ö.A., P.Y.G., M.F.E., E.G.B.; Tasarım ve Dizayn -

Ö.A., P.Y.G., M.F.E., E.G.B.; Denetleme - Ö.A., P.Y.G.,

M.F.E., E.G.B.; Kaynaklar - Ö.A.; Malzemeler - Ö.A.;

Veri Toplama ve/veya İşleme - Ö.A.; Analiz ve/veya Yorum

- Ö.A., P.Y.G.; Literatür Taraması - Ö.A., P.Y.G.;

Yazıyı Yazan - Ö.A.; Eleştirel İnceleme - Ö.A.

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5. Yang Q, Rasmussen SA, Friedman J. Mortality associated

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8. Bertrand P, Navarro H, Caussade S, Holmgren N,

Sánchez I. Airway anomalies in children with Down syndrome:

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Pneumonia, lung abscess, and empyema. Semin Pediatr

Surg 2008; 17:42-52. [CrossRef]

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abscess. Surg Infect (Larchmt) 2013; 14:335-6.

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what is optimal therapy. Infect Dis Clin North Am 2013;

27:149-55. [CrossRef]

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JD, Silvestri RC, Koziel H. Lung abscess in adults: clinical

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patients. Respir Med 2002;

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ML, McCarron B, et al. Pyogenic brain abscess and subdural

empyema: presentation, management, and factors

predicting outcome. Infection 2018; 46:785-92.

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15. Karageorgiou I, Chandler C, Whyte MB. Silent diabetes

mellitus, periodontitis and a new case of thalamic abscess.

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with Down syndrome. Dent Clin North Am 2016;

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S. Abcès du poumon : diagnostic et prise en

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18. Bokhari MR, Mesfin FB. Brain Abscess. In: StatPearls,

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a challenge that Magnetic Resonance can help us

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20. Radoi M, Ciubotaru V, Tataranu L. Brain abscesses: clinical

experience and outcome of 52 consecutive cases.

Chirurgia (Bucur) 2013; 108(2):215-25.



OLGU SUNUMU

CASE REPORT

A Case of Primary Pleural Synovial Sarcoma

Primer Plevral Sinovyal Sarkom Olgusu

Kadir Canoglu 1 , Ismail Yilmaz 2 , Tayfun Caliskan 1 , Omer Ayten 1 , Oguzhan Okutan 1 ,

Zafer Kartaloglu 1 , Neslihan Kaya Terzi 3


Abstract

We present here the case of a 29-year-old male with

primary pleural synovial sarcoma. The patient had no

complaints, no significant medical history and did not

smoke. A well-circumscribed giant mass, adjacent to

the chest wall was observed on a PA chest X-ray, with

a solid mass lesion in the lateral right hemithorax with

diffuse amorphous calcifications noted on thorax

computed tomography. An F18-FDG uptake with a

SUVmax of 5.0 was detected in the mass defined by

PET/CT. A Tru-cut biopsy revealed monophasic as

histological subtypes composed of a dense cellular

and interlaced fascicular proliferation of spindle cells,

and SYT-SSX1 fusion (X;18)(p11.23;q11) consistent

with synovial sarcoma. The patient was followed-up

in the oncology center after surgery and chemoradiotherapy,

and has been stable for 1 year. This paper

draws attention to a diagnostic approach to primary

pleural synovial sarcoma, which is a rare and aggressive

tumor, and the need for the prompt initiation of

treatment for an improved outcome.

Key words: Lung cancer, pleura, synovial sarcoma.

1 Department of Pulmonology, Health Science University, Sultan II.

Abdülhamid Han Training and Research Hospital, İstanbul,

Türkiye

2 Department of Pathology, Health Science University, Sultan II.

Abdülhamid Han Training and Research Hospital, İstanbul,

Türkiye

3 Department of Pathology, Health Science University,

Gaziosmanpaşa Training and Research Hospital, İstanbul, Türkiye

Öz

Primer plevral sinovyal sarkom tanılı 29 yaşında erkek

hastanın, gelişinde herhangi bir şikayeti yoktu, sigara

içmiyordu ve özgeçmişinde özellik yoktu. PA akciğer

grafisinde, göğüs duvarına bitişik düzgün sınırlı dev

kitle izlendi. Toraks BT’sinde; sağ hemitoraks lateralinde,

diffüz amorf kalsifikasyon içeren solid kitlesel

lezyon tespit edildi. PET/BT’de, tanımlanan lezyonda

SUVmax 5.0 olan F18-FDG tutulumu saptandı. Trucut

biyopsi sonucunda, histolojik olarak monofazik,

yoğun hücresel ve birbiri üzerine geçen fasiküllerin

oluşturduğu iğsi hücreler, SYT-SSX1 füzyon

(X;18)(p11.23;q11) saptanması üzerine sinovyal

sarkom tanısı kondu. Cerrahi ve kemoradyoterapi

sonrasında 1 yıldır stabil olarak onkoloji merkezinde

takip edilmektedir. Bu yazı, nadir görülen ve agresif

bir tumor olan primer plevral sinovyal sarkomun tanı

basamakları ve iyi sonuçlar alınabilmesi için hızlıca

tedavisinin başlanmasına dikkat çekmeyi amaçlamaktadır.

Anahtar Sözcükler: Akciğer kanseri, plevra, sinovyal

sarkom.

1 Sağlık Bilimleri Üniversitesi, Sultan 2. Abdülhamid Han

Eğitim ve Araştırma Hastanesi, Göğüs Hastalıkları Kliniği,

İstanbul

2 Sağlık Bilimleri Üniversitesi, Sultan 2. Abdülhamid Han

Eğitim ve Araştırma Hastanesi, Patoloji Kliniği, İstanbul

3 Sağlık Bilimleri Üniversitesi, Gaziosmanpaşa Eğitim ve

Araştırma Hastanesi, Patoloji Kliniği, İstanbul


Correspondence (İletişim): Kadir Canoglu, Department of Pulmonology, Health Science University, Sultan II. Abdülhamid Han Training

and Research Hospital, İstanbul, Türkiye

e-mail: kadircano@gmail.com

88


Although synovial sarcoma is relatively rare, it accounts

for 5–10% of all soft tissue sarcomas. It is frequently seen

in adolescents and young adults, and is diagnosed equally

in both sexes (1). It is often found in the lower extremities,

and primarily in the hip or knee-joint regions in periarticular

soft tissue, and can rarely be of bone origin (2).

Solid tumors of pleural origin other than malignant mesothelioma

include desmoid type fibromatosis, neurogenic

tumors, liposarcoma, epithelioid hemangioendothelioma,

solitary fibrous tumors and synovial sarcoma (3). Primary

synovial sarcoma is very rare in the lung, accounting for

0.5% of all lung cancers (4). Fewer than 50 cases of

pleural synovial sarcoma have been reported in literature

(5). Immunohistochemical staining for transducine-like

enhancers of split 1 (TLE1), cytokeratins (CK), epithelial

membrane antigens (EMA), PANCK (cytokeratin

AE1/AE3), B cell lymphoma 2 (BCL2), beta catenin, calponin,

CD 56 (cluster of differentiation), CD57, CD99

and calretinin and SS18-SSX fusion-specific antibodies

are used for definitive diagnosis (6). Here, we present the

case of a 29-year-old male with primary pleural synovial

sarcoma.

CASE

A 29-year-old male patient was referred to our outpatient

clinic after a lesion was observed in a PA chest X-ray

taken for a workplace health check-up. The patient had

no complaints and did not smoke. He had no significant

disease history and no comorbid disease. White blood

count: 10980 mm 3 , hemoglobin: 14.5 g/dL, hematocrit:

42.9%, platelet count: 291000 mm 3 , C-reactive protein

(CRP): 33.3 mg/L, international normalized ratio (INR):

1.12, lactate dehydrogenase: 405 U/L and creatinine:

1.05 mg/dL were detected. There was no pathology in a

postero-anterior (PA) chest X-ray taken 3 years earlier. A

well-circumscribed giant mass, adjacent to the chest wall,

forming a wide angle with the chest wall, almost completely

filling the upper and middle zone of the right lung

was observed in the current PA chest X-ray (Figure 1). A

10x13x14 cm solid mass lesion in the lateral right hemithorax

with its wide base sitting on the pleura and a

regular free contour causing compression on the lung

and with diffuse amorphous calcifications was detected

on thorax computed tomography (CT) (Figure 2). No

mediastinal or hilar lymph nodes were observed on CT.

Heterogeneous fluorine-18-fluorodeoxyglucose (F18-

FDG) uptake with a standardized uptake value (SUV) max

of 5.0 was detected in the mass, defined from positron

emission tomography (PET)/CT taken for metastasis evaluation,

and pathological FDG uptake with a SUVmax of

5.0 was detected in the 1 cm parenchymal nodular lesion

posterior to this lesion and adjacent to mild pleural effusion.

There was no other primary focus other than the

lung on PET/BT. Ultrasound guided tru-cut biopsy was

performed on the giant mass through interventional radiology.

Monophasic histological subtypes composed of

dense cellular and interlacing fascicular proliferations of

spindle cells were seen. CK7 (focal), CK19 (focal), beta

catenin, TLE1 and CD99 were positive; SRY-related

HMG-box 10 (SOX10), calretinin, Wilm's tumor 1 (WT1),

S100 protein, CD34, CD117, Desmin, smooth muscle

actin (SMA) and human melanoma black-45 (HMB45)

were negative in immunohistochemical (IHC) staining,

and the Ki67 proliferation index was 10% (Figure 3).

Rearrangement was detected in the SS18 (SYT) gene by a

fluorescence in-situ hybridization (FISH) technique. Fusion

of the SYT gene located on chromosome 18 to the synovial

sarcoma X-1 (SSX1) gene in the Xp11 region (SYT-

SSX1 fusion) was detected in an investigation of reciprocal

chromosomal translocations (X;18)(p11.23;q11)

using a reverse transcriptase-polymerase chain reaction

(RT-PCR) technique.

A diagnosis of synovial sarcoma was made based on the

available histopathological findings. The patient was

referred to surgery, but opted to undergo surgery in another

thoracic surgery clinic unconnected to our hospital.

The patient’s medical records revealed that he underwent

a mass excision and visceral and parietal decortication,

and since no palpable nodule was detected during surgery,

no lung parenchyma resection was performed. A

postoperative control PET/CT revealed that the parenchymal

nodule had regressed spontaneously, as well as

the pleural effusion. However, since there was suspicion

about R0 resection and a 1 cm sized nodule was detected

in the lower lobe, the patient was referred to medical

oncology for chemoradiotherapy. The mitosis rate was

lower than 10/10 high-power field (HPF), and the necrosis

rate was less than 50%.

DISCUSSION

Primary pulmonary synovial sarcoma is a very rare, highly

aggressive malignant tumor that is detected equally in

both sexes and mostly in young adults, and accounts for

0.5% of all lung cancers (7). The METASARC study reported

5-year survival in only 8.5% (16/188) of patients

with a diagnosis of metastatic synovial sarcoma following

diagnosis (8). Clinical features, radiological findings,

histopathological features and genetic parameters are


A Case of Primary Pleural Synovial Sarcoma | Canoglu et al.

important in the diagnosis and differential diagnosis of

synovial sarcoma, which is a rare disease associated with

high mortality.

Although primary pulmonary synovial sarcoma can involve

any lobe radiologically, it is reported mostly in the

upper lobes, and can be located centrally or peripherally

in the lung. Chest pain is the most common complaint, as

well as symptoms of cough, hemoptysis, weight loss, fever,

and shortness of breath. Patients may also be completely

asymptomatic, with radiological masses detected only

incidentally (9,10). In the present case, the radiological

lesion was detected incidentally in a non-smoking asymptomatic

young adult patient.

There was no radiological finding of calcification in Hartel

et al.’s (11) primary pulmonary synovial sarcoma study

of 60 patients, while diffuse amorphous calcification

areas were observed in our case. Concurring with literature,

a mild F18-FDG uptake was detected in the mass

lesion in our case. In the study by Lan et al. (12), in four

of the 26 patients studied, the pleurapulmonary and mediastinal

synovial sarcoma were of pleural origin The

authors also reported detecting an ipsilateral pleural

effusion in 12 of 21 (57.1%) patients. In our case, an

ipsilateral pleural effusion was detected at the time of

diagnosis, and complete regression was observed postoperatively.

Figure 2: A 10x13x14 cm solid mass lesion in the lateral right hemithorax

with its wide base sitting on the pleura and with a regular free

contour, causing compression in the lung and with diffuse amorphous

calcifications on thorax computed tomography

Figure 3: Histologic features of synovial sarcoma. Synovial sarcoma is

one of the most cellular spindle-cell neoplasms of the lung, and is characteristically

composed of densely cellular interlacing fascicles (A).

Tumor cells showing diffuse nuclear staining with TLE-1 (B). Cytokeratin

positivity is critical for diagnosis and can be minimal (C). Moderate

intensity CD99 staining (D) and strong beta-catenin staining have shown

(E). Slightly high Ki-67 proliferative index (F)

Figure 1: A well-circumscribed giant mass adjacent to the chest wall

forming a wide angle with the chest wall, almost completely filling the

upper and middle zone of the right lung on a PA chest X-ray

Soft tissue tumors present with quite different clinics due

to their histological differences, variable pathological

findings and genetic changes. The histological subtypes

of synovial sarcoma are monophasic, biphasic and poorly

differentiated. Among these, the synovial sarcoma monophasic

subtype can be confused with other soft tissue

tumors, and so IHC examinations are important in a

differential diagnosis (13,14). The monophasic subtype

was detected histologically in this case report.



The combination of EMA and cytokeratin positivity and

CD34 negativity has been stated as the most useful

marker in the diagnosis of synovial sarcoma in IHC staining

(15). In the presented case, CK7, CK19, PANCK,

beta catenin, TLE1 and CD99 were positive; and SOX10,

calretinin, WT1, S100 protein, CD34, CD117, desmin,

SMA and HMB45 were negative. The optimum approach

to the definitive diagnosis of synovial sarcoma is the

demonstration of SS18-SSX (2/3 SSX1, 1/3 SSX2 and

rarely SSX4) fusion by RT-PCR or FISH techniques. In a

recent study investigating the diagnostic efficacy of the

SS18-SSX fusion-specific antibody, 93 masses, including

10 synovial sarcomas metastasizing to the lung, and five

intrathoracic solitary fibrous tumors, 39 primary lung

cancers, and 49 non-synovial sarcomas metastasizing to

the lung were compared. While SS18-SSX was positive in

all 10 synovial sarcomas, it was negative in all 93 nonsynovial

sarcoma masses (100% sensitivity, 100% specificity)

(16). A definitive diagnosis was made by demonstrating

SS18-SSX1 fusion with the FISH technique in the

presented case.

Although there is no clear recommendation for optimal

treatment, the leading treatment option is surgery, involving

the complete removal of the mass, lobectomy or

pneumonectomy. Synovial sarcomas are relatively

chemotherapy-sensitive tumors. Adriamycin alone or in

combination with ifosfamide can be used, while radiotherapy

can be used for the local control of the disease

(11,17). In the presented case, tumor excision and decortication

were performed, and the patient was then referred

to oncology for chemoradiotherapy.

Advanced age, female gender, R1 and R2 resection,

tumor size >5 cm, extensive tumor necrosis, high mitotic

activity and neurovascular invasion have been reported

as poor prognostic criteria. Although SYT-SSX1 fusion has

also been stated to be a poor prognostic factor, other

studies reported opposite findings (4,18). The present

case was a 29-year-old male with a >5 cm tumor, an

unclear R0 resection, mild tumor necrosis and low mitotic

activity, and SYT-SSX1 fusion was detected. The patient

has maintained a stable condition for 1 year on follow-up

in the oncology center.

CONCLUSION

Primary synovial sarcoma is very rare in the lung, and is a

highly aggressive malignant tumor that is detected equally

in both sexes, and mostly in young adults. We presented

this case to draw attention to the approaches to the diagnosis

and treatment of this rare disease.


None declared.


Concept - K.C., I.Y., T.C., O.A., O.O., Z.K., N.K.T.;

Planning and Design - K.C., I.Y., T.C., O.A., O.O., Z.K.,

N.K.T.; Supervision - K.C., I.Y., T.C., O.A., O.O., Z.K.,

N.K.T.; Funding -; Materials - K.C., I.Y., T.C., O.A.,

N.K.T.; Data Collection and/or Processing - K.C., I.Y.,

T.C., O.A., N.K.T.; Analysis and/or Interpretation - K.C.,

I.Y., T.C., O.A., N.K.T.; Literature Review - K.C., T.C.,

O.O., Z.K., N.K.T.; Writing - K.C., T.C., O.A., O.O.,

Z.K.; Critical Review - K.C., I.Y., T.C., O.A., O.O., Z.K.,

N.K.T.

YAZAR KATKILARI

Fikir - K.C., I.Y., T.C., O.A., O.O., Z.K., N.K.T.; Tasarım

ve Dizayn - K.C., I.Y., T.C., O.A., O.O., Z.K., N.K.T.;

Denetleme - K.C., I.Y., T.C., O.A., O.O., Z.K., N.K.T.;

Kaynaklar -; Malzemeler - K.C., I.Y., T.C., O.A., N.K.T.;

Veri Toplama ve/veya İşleme - K.C., I.Y., T.C., O.A.,

N.K.T.; Analiz ve/veya Yorum - K.C., I.Y., T.C., O.A.,

N.K.T.; Literatür Taraması - K.C., T.C., O.A., O.O., Z.K.;

Yazıyı Yazan - K.C., T.C., O.O., Z.K., N.K.T.; Eleştirel

İnceleme - K.C., I.Y., T.C., O.A., O.O., Z.K., N.K.T.

REFERENCES

1. Gazendam AM, Popovic S, Munir S, Parasu N, Wilson D,

Ghert M. Synovial sarcoma: a clinical review. Curr Oncol

2021; 28:1909-20. [CrossRef]

2. Caracciolo JT, Henderson-Jackson E, Binitie O. Synovial

sarcoma of bone: Sarcoma typically of soft tissues presenting

as a primary bone tumor. Radiol Case Rep 2018;


3. Attanoos RL, Pugh MR. The diagnosis of pleural tumors

other than mesothelioma. Arch Pathol Lab Med 2018;

142:902-13. [CrossRef]

4. Pandey L, Joseph D, Pasricha R, Gupta MK. Primary synovial

sarcoma of the lung: a rare presentation, diagnostic

dilemma and review of literature. BMJ Case Rep 2020;


5. Jiang M, Zhang J, Cheng D. A second primary synovial

sarcoma of pleural developed nine-years after the first

synovial sarcoma of plantar pedis: a case report and review

of literature. Int J Clin Exp Pathol 2019; 12:2743-8.

6. Obeidin F, Alexiev BA. Synovial sarcoma. PathologyOutlines.com

website. Access date: 20 October 2021.

Available

at:


A Case of Primary Pleural Synovial Sarcoma | Canoglu et al.

http://www.pathologyoutlines.com/topic/softtissuesynovia

lsarc. html.

7. Pereira G, Pires A, Barbosa S, Cotter J. Pulmonary synovial

sarcoma: an unpleasant purprise. Eur J Case Rep Intern

Med 2021; 8:002149. [CrossRef]

8. Savina M, Le Cesne A, Blay JY, Ray-Coquard I, Mir O,

Toulmonde M, et al. Patterns of care and outcomes of

patients with METAstatic soft tissue SARComa in a reallife

setting: the METASARC observational study. BMC

Med 2017; 15:78. [CrossRef]

9. Zeren H, Moran CA, Suster S, Fishback NF, Koss MN.

Primary pulmonary sarcomas with features of monophasic

synovial sarcoma: a clinicopathological, immunohistochemical,

and ultrastructural study of 25 cases. Hum

Pathol 1995; 26:474-80. [CrossRef]

10. Etienne-Mastroianni B, Falchero L, Chalabreysse L, Loire

R, Ranchère D, Souquet PJ, et al. Primary sarcomas of

the lung: a clinicopathologic study of 12 cases. Lung

Cancer 2002; 38:283-9. [CrossRef]

11. Hartel PH, Fanburg-Smith JC, Frazier AA, Galvin JR,

Lichy JH, Shilo K, et al. Primary pulmonary and mediastinal

synovial sarcoma: a clinicopathologic study of 60

cases and comparison with five prior series. Mod Pathol

2007; 20:760-9. [CrossRef]

12. Lan T, Chen H, Xiong B, Zhou T, Peng R, Chen M, et al.

Primary pleuropulmonary and mediastinal synovial sarcoma:

a clinicopathologic and molecular study of 26 genetically

confirmed cases in the largest institution of

southwest China. Diagn Pathol 2016; 11:62. [CrossRef]

13. Stamenovic D, Hohenberger P, Roessner E. Pulmonary

metastasectomy in soft tissue sarcomas: a systematic review.

J Thorac Dis 2021; 13:2649-60. [CrossRef]

14. Okamoto S, Hisaoka M, Daa T, Hatakeyama K, Iwamasa

T, Hashimoto H. Primary pulmonary synovial sarcoma:

a clinicopathologic, immunohistochemical, and molecular

study of 11 cases. Hum Pathol 2004; 35:850-6.

[CrossRef]

15. Bégueret H, Galateau-Salle F, Guillou L, Chetaille B,

Brambilla E, Vignaud JM, et al. Primary intrathoracic synovial

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16. Miura K, Shimizu K, Eguchi T, Koike S, Matsuoka S,

Takeda T, et al. Usefulness of SS18-SSX antibody as a

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Diagn Pathol 2021; 16:54. [CrossRef]

17. Shah S, Sankrithi P, Shah K, Dalia S, Rudrappa M. Primary

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Cureus 2020; 12:e11899. [CrossRef]

18. Guillou L, Benhattar J, Bonichon F, Gallagher G, Terrier

P, Stauffer E, et al. Histologic grade, but not SYT-SSX fusion

type, is an important prognostic factor in patients

with synovial sarcoma: a multicenter, retrospective analysis.

J Clin Oncol 2004; 22:4040-50. [CrossRef]



OLGU SUNUMU

CASE REPORT

Kommerell Diverticulum: A Rare Cause of

Dyspnea and Dysphagia

Dispne ve Disfajinin Nadir Bir Nedeni Olarak Kommerell Divertikülü

Refika Hural 1 , Gülsüm Bingol 3 , Enis Öztürk 2 , Ebru Serin 4 , Seda Tural Onur 5 ,

Emir Özgür Barış Ökçün 3


Abstract

Kommerell's diverticulum (KD) is a congenital dilatation

of the distal portion of the aortic arch that is

usually located at the origin of an aberrant right

subclavian artery (ARSA) or the aberrant left subclavian

artery (ALSA). We describe here a 40-year-old

male patient with a right-sided aortic arch (RAA) with

ligamentum arteriosum (LA) and ALSA originating

from the KD. The applied to our outpatient clinic with

a history of dyspnea and dysphagia that had persisted

for the last year. The patient underwent chest

computed tomography (CT) in our clinic, revealing

ALSA-level KD in RAA with ligamentum arteriosum.

Surgical resection of KD and LA was planned based

on the symptoms. NE was resected from the descending

aorta and ALSA was transferred to the left carotid

artery, and the patient was discharged 6 days after

the operation without complications. After the operation,

the patient's dyspnea and dysphagia subsided

completely.

Key words: Kommerell Diverticulum, Congenital

Anomalies, ARSA, ALSA.

1 Department of Cardiology, Dr. Burhan Nalbantoğlu State

Hospital, Nicosia, Cyprus

2 Bakırköy Prof. Mazhar Osman Research and Training Hospital for

Neurologic and Psychiatric Diseases, İstanbul, Türkiye

3 Department of Cardiology, Memorial Bahçelievler Hospital,

İstanbul, Türkiye

4 Department of Cardiology, İstanbul University Cerrahpaşa,

Institude of Cardiology, İstanbul, Türkiye

5 Department of Chest Diseases, Yedikule Chest Diseases and T

horacic Surgery Training and Research Hospital, İstanbul, Türkiye

Öz

Kommerell divertikülü (KD), genellikle aberran bir sağ

subklavyen arterin (ARSA) veya aberran sol subklavyen

arterin (ALSA) çıkışında bulunan aortik arkın distal

kısmının konjenital dilatasyonudur. Bu olguda, sağ

taraflı aortik ark (RAA) ile beraber ligamentum arteriyozumu

(LA) olan ve KD'den kaynaklanan ALSA'sı

olan 40 yaşında bir erkek hastayı tanımladık. Polikliniğimize

başvurmuş bu hastanın son bir yıl içinde

devam eden nefes darlığı ve yutma güçlüğü öyküsü

vardı. Kliniğimizde hastaya, ligamentum arteriyozumlu

RAA'da ALSA düzeyinde KD’ü göstermek için akciğer

tomografisi çekildi. Semptomlar nedeniyle KD ve

LA’nın cerrahi rezeksiyonu planlandı. İnen aortadan

KD rezeke edildi ve ALSA sol karotid artere transfer

edildi. Hasta ameliyattan 6 gün sonra komplikasyonsuz

olarak taburcu edildi. Ameliyattan sonra hastanın

dispne ve disfajisi tamamen kayboldu.

Anahtar Sözcükler: Kommerell Divertikülü, Konjenital

Anomaliler, ARSA, ALSA.

1 Dr. Burhan Nalbantoğlu Devlet Hastanesi, Kardiyoloji

Bölümü, Lefkoşa, Kıbrıs

2 Bakırköy Prof. Dr. Mazhar Osman Ruh Sağlığı ve Sinir

Hastalıkları Eğitim ve Araştırma Hastanesi, Radyoloji Anabilim

Dalı, İstanbul

3 Memorial Bahçelievler Hastanesi, Kardioloji Bölümü, İstanbul

4 İstanbul Üniversitesi Cerrahpaşa, Kardiyoloji Enstitüsü,

Kardioloji Bölümü, İstanbul

5 Yedikule Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim

Araştırma Hastanesi, Göğüs Hastalıkları Bölümü, İstanbul


Correspondence (İletişim): Gülsüm Bingol, Department of Cardiology, Memorial Bahçelievler Hospital, İstanbul, Türkiye

e-mail: bulut_gulsum@hotmail.com

93


Kommerell diverticulum (KD) is congenital dilatation of

the distal part of the aortic arch that is usually found at

the origin of an aberrant right subclavian artery (ARSA) or

aberrant left subclavian artery (ALSA) (1). It was first described

in 1937 by B. F. Kommerell, whose definition

related to a patient with ARSA and left aortic arch (LAA)

(2). Although most patients with KD are asymptomatic,

dilatation of the diverticulum may compress the trachea

or esophagus, causing such symptoms as dysphagia,

dyspnea, wheezing, coughing and chest pain. The presence

of an abnormal subclavian artery or vascular ring

may worsen compression symptoms (2,3). Esophageal

and tracheal occlusions are well-known indications for

the surgical treatment of KD (4).

CASE

We describe here the case of a 40-year-old man who

was admitted to our outpatient clinic with a right-sided

aortic arch (RAA) with a ligamentum arteriosum (LA) and

an ALSA arising from a KD. The patient had a history of

dyspnea and dysphagia lasting for the last one year, as

well as hypertension and diabetes mellitus. A physical

examination revealed blood pressure of 120/70 mmHg

that was the same in both arms, and weak left brachial

and radial artery pulses.

A chest X-ray revealed an enlarged artery in the proximal

descending aorta. Laboratory results were within normal

limits. Transthoracic echocardiography showed normal

left ventricular systolic function. CT was performed to

demonstrate KD at the level of the ALSA in the RAA with a

ligamentum arteriosum (Figure 1). The origin of ALSA,

which causes significant compression by the LA (not

demonstrated in the figures) and RAA in both the esophagus

and trachea, has been shown to be aneurysmal

(Figure 1 and 2).

Surgical resection of the KD and LA was planned based

on the symptoms. The LA between the origin of the descending

aorta and the left pulmonary artery was separated.

The KD was resected from the descending aorta

and the ALSA was transferred to the left carotid artery.

The patient was discharged 6 days after the operation

without complication. The dyspnea and dysphagia subsided

completely after the operation. Postoperative CT

images are presented in Figures 3, 4 and 5.

Figure 1: Axial CT (a) and corresponding magnetic resonance (MR)

images (b) show the RAA and KD causing mild compression of the

trachea and prominent compression of the esophagus

Figure 2: Coronal CT showing the ALSA arising from KD

DISCUSSION

The prevalence of congenital aortic arch anomalies

ranges from 1–2% in the general population (5). These

anomalies are commonly asymptomatic, and can be

left/right-sided, double (DAA) or cervical aortic arch

(CAA). Regardless of which, abnormal isolations of the


Kommerell Diverticulum: A Rare Cause of Dyspnea and Dysphagia | Bingol et al.

subclavian, brachiocephalic or carotid arteries and vascular

rings may produce symptoms (6).

A normal LAA is generated by the regression of the right

canal, RAA and the right dorsal aorta. The first branch of

a normal LAA is the right brachiocephalic artery, followed

by the left common carotid and left subclavian arteries.

Furthermore, the first branch of a normal LAA is the right

brachiocephalic artery, followed by the left common carotid

and left subclavian arteries. The prevalence of RAAs

ranges from 0.05–0.1% of the population (7), while

DAAs and CAAs are rarer vascular anomalies.

According to the branching pattern of great vessels referred

to by Bravo et al. (5), the LAA can divide into three

groups as ARSA, right subclavian artery isolation and

normal. Furthermore, RAAs can be divided into three

types based on the branching pattern of the arch veins as

left subclavian artery isolation, mirror image branching

and ALSA (6).

In the study by Türkvatan et al. (8), one of the most common

congenital anomalies of the aortic arch was stated

to be aberrant subclavian artery, whereas the incidence

of ARSA resulting from normal LAA is in the 0.4–2.3%

range in the general population. Yang et al. (9) reported

that ALSA originating from the RAA is rare, with an incidence

of only 0.05% in the general population.

KD is an aneurysmal dilatation of the distal part of the

aortic arch or at the origin of an aberrant subclavian

artery (4). KD is widely thought to result from degeneration

associated with atherosclerosis, or to be congenital

(10). It has also been reported that this condition, which

is more common after the age of 50, has no gender

predominance. According to Kommorell's initial definition

(2), the KD consists of an abnormal artery originating

from the left aortic arch, while the right subclavian artery

emerges as the last branch of the aortic arch. This branch

then passes from the proximal descending aorta behind

the esophagus to the right arm.

Figure 3: Post-operative coronal CT image showing metallic clips at the

site of the resected KD

Figure 4: Postoperative coronal CT image showing a carotico-subclavian

by-pass

Figure 5: Postoperative axial CT images showing a carotico-subclavian

by-pass



Although the existence of seven types of KD has been

reported in literature, three are more common: (a) LAA

diverticula with ARSA, (b) RAA diverticula with ALSA, and

(c) diverticula at the aortic-ductal junction (4,8). Patients

are usually asymptomatic, but may experience tracheoesophageal

compression, although dilation of the KD

can sometimes lead to such symptoms as shortness of

breath, wheezing, coughing or chest pain due to the

compression of the trachea or esophagus (2,4,11).

Vascular rings are rare congenital anomalies of the aortic

arch and its branches in which the trachea and esophagus

may be compressed by a combination of LA and an

abnormal aortic arch course. The RAA is the second most

common form of a complete vascular ring formed by a

patent duct or ligamentum arteriosum, contralateral to

the ascending aorta (12). Different imaging methods,

such as echocardiography, barium swallow, bronchoscopy,

esophagography, angiography, CT and magnetic

resonance imaging (MRI) are available for the diagnosis

of this anomaly (11). CT and MRI are considered the best

diagnostic approaches to KD. The diagnosis of our case

was confirmed by CT angiography, which shows the RAA

with the ALSA and KD.

Complications of KD can be serious, with diverticulum

rupture or dissection reported in patients with aberrant

subclavian arteries (13). Symptomatic patients should be

considered candidates for surgery when the minimum size

of the KD is greater than 2 cm or the aneurysm diameter

exceeds 1.5 times that of the associated subclavian artery

surgery (14).

Although surgical interventions in asymptomatic patients

is controversial, some authors recommend surgery in such

patients due to the risk of rupture or dissection (3). Various

surgical techniques are available in this regard, such

as simple ligation of the left subclavian artery, resection

of the KD, transplantation of the left subclavian artery to

the left carotid artery, and endovascular repair (4,11).


None declared.


Concept - R.H., G.B., E.Ö., E.S., S.T.O., E.Ö.B.Ö.;

Planning and Design - R.H., G.B., E.Ö., E.S., S.T.O.,

E.Ö.B.Ö.; Supervision - R.H., G.B., E.Ö., E.S., S.T.O.,

E.Ö.B.Ö.; Funding -; Materials - R.H., E.Ö.; Data Collection

and/or Processing - R.H., E.Ö.; Analysis and/or

Interpretation - G.B., S.T.O.; Literature Review – E.S.,

G.B.; Writing - G.B., R.H., E.Ö.B.Ö.; Critical Review -

S.T.O., E.Ö.B.Ö., G.B.

YAZAR KATKILARI

Fikir - R.H., G.B., E.Ö., E.S., S.T.O., E.Ö.B.Ö.; Tasarım

ve Dizayn - R.H., G.B., E.Ö., E.S., S.T.O., E.Ö.B.Ö.;

Denetleme - R.H., G.B., E.Ö., E.S., S.T.O., E.Ö.B.Ö.;

Kaynaklar -; Malzemeler - R.H., E.Ö.; Veri Toplama

ve/veya İşleme - R.H., E.Ö.; Analiz ve/veya Yorum - G.B.,

S.T.O.; Literatür Taraması - E.S., G.B.; Yazıyı Yazan -

G.B., R.H., E.Ö.B.Ö.; Eleştirel İnceleme - S.T.O.,

E.Ö.B.Ö., G.B.

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diverticulum and aneurysmal right-sided aortic arch: A

case report and review of the literature. J Vasc Surg 2000;

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and Kommerell's diverticulum. Tex Heart Inst J 2002;

29:109-12.

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of Kommerell diverticulum. Can Assoc Radiol J

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the current era: a comprehensive review. Gen Thorac

Cardiovasc Surg 2015; 63:245-59. [CrossRef]

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Fetal aortic arch anomalies: key sonographic views for

their differential diagnosis and clinical implications using

the cardiovascular system sonographic evaluation protocol.

J Ultrasound Med 2016; 35:237-51. [CrossRef]

6. Stojanovska J, Cascade PN, Chong S, Quint LE,

Sundaram B. Embryology and imaging review of aortic

arch anomalies. J Thorac Imaging 2012; 27:73-84.

[CrossRef]

7. Hastreiter AR, D'Cruz IA, Cantez T. Right-sided Aorta. Br

Heart J 1966; 28:722-39. [CrossRef]

8. Türkvatan A, Büyükbayraktar FG, Ölçer T, Cumhur T.

Congenital anomalies of the aortic arch : evaluation with

the use of multidetector. Korean J Radiol 2009; 10:176-

84. [CrossRef]

9. Yang C, Shu C, Li M, Li Q, Kopp R. Aberrant subclavian

artery pathologies and Kommerell's diverticulum : a review

and analysis of published endovascular/hybrid

treatment options. J Endovasc Ther 2012; 19:373-82.

[CrossRef]


Kommerell Diverticulum: A Rare Cause of Dyspnea and Dysphagia | Bingol et al.

10. Faggioni L, Gabelloni M, Napoli V, Iorio F, Chella A,

Caramella D. Thrombosis of Kommerell's diverticulum

with subclavian steal phenomenon in a patient with nonsmall

cell lung carcinoma under chemotherapy. Eur J

Radiol Open 2016; 3:191-4. [CrossRef]

11. Tashnizi MA, Ghorbanzadeh A, Zirak N, Hoseinikhah H,

Moeinipour A. Aberrant left subclavian artery associated

with Kommerell diverticulum causing severe nausea and

vomiting : a case report and review of literature. Iran

Hear J 2018; 19:71-4.

Seminars in Pediatric Surgery. Elsevier; 2016: 1-40.

[CrossRef]

13. Idrees J, Keshavamurthy S, Subramanian S, Clair DG,

Svensson LG, Roselli EE. Hybrid repair of Kommerell diverticulum.

J Thorac Cardiovasc Surg 2014; 147:973-6.

[CrossRef]

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Surgical treatment for Kommerell's diverticulum. J Thorac

Cardiovasc Dis 2006; 131:574-8. [CrossRef]

12. Backer CL, Mongé MC, Andrada R, Eltayeb OM, Rastatter

JC, Rigsby CK. Vascular rings. In: Grosfeld JL, edt.



OLGU SUNUMU

CASE REPORT

Incidentally Detected Aberrant Right

Subclavian Artery: A Case Report

İnsidental Tespit Edilen Aberran Sağ Subklavyen Arter: Olgu Sunumu

Mehmet Ağar,

Semih Koçyiğit


Abstract

Aberrant right subclavian artery (ARSA) is a rarely

seen congenital and often asymptomatic anomaly.

The most common clinical presenting symptom in

adult ARSA patients is dysphagia, while patients may

rarely present with respiratory complaints. We present

here the case of a 79-year-old patient with dysphagia

who was diagnosed based on thorax and neck CT

and esophagography findings. The patient was treated

for pneumonia in the centers to which she applied

with complaints of cough and inability to swallow,

and was subsequently referred to us with the suspicion

of a mediastinal mass after radiograms were

obtained. The diagnosis of ARSA was made based on

contrast-enhanced computed tomography. With this

in mind, ARSA should be included in the differential

diagnosis of patients presenting with dysphagia,

despite it being a rarely seen etiology.

Key words: Right subclavian artery, esophagus, dysphagia.

Öz

Aberran sağ subklavyen arter (ASSA) konjenital bir

anomali olup, sıklıkla asemptomatik seyreden ve

nadir görülen bir anomalidir. Erişkin ASSA’lı hastalarda

en sık klinik başvuru semptomu disfaji olup

nadirde olsa solunum yakınmaları ile de başvurabilirler.

Yutma güçlüğü ile başvuran 79 yaşındaki bir

hasta çekilen toraks ve boyun BT, özofagografi ile

tanı konularak sunulmuştur. Hasta yutkunamama ve

öksürük şikayeti ile başvurduğu merkezlerde pnömoni

tedavisi görmüş, çekilen grafiler sonrası mediastinal

kitle şüphesiyle sevk edilen hastaya kontrastlı bilgisayarlı

tomografi ile ASSA tanısı konulmuştur. Sonuç

olarak yutma güçlüğü ile başvuran hastalarda ASSA

buna neden olan nadir bir sebepte olsa ayırıcı tanıda

yer almalıdır.

Anahtar Sözcükler: Sağ subklavyen arter, özofagus,

disfaji.

Department of Thoracic Surgery, Fethi Sekin City Hospital, Elazığ,

Türkiye

Fethi Sekin Şehir Hastanesi, Göğüs Cerrahi Kliniği, Elazığ


Correspondence (İletişim): Mehmet Ağar, Department of Thoracic Surgery, Fethi Sekin City Hospital, Elazığ, Türkiye

e-mail: md.mehmetagar@gmail.com

98


ARSA was first described in an autopsy performed by

David Bayford (1) in 1794. It is a congenital anomaly that

often courses asymptomatically without any clinical findings,

and is the most common aortic arch anomaly seen

in autopsy series, with a prevalence of 2.5% (2).

Diagnoses of ARSA, which mostly leads an asymptomatic

course, are often made incidentally, and mostly through

imaging methods performed for such complaints as dysphagia,

cough and stridor resulting from the aberrant

artery compressing surrounding tissues. Dysphagia is the

most common among these symptoms, and was for many

years referred to as “dysphagia lusoria”.

Figure 1: The patient's PA AC was unremarkable. A non-contrast thorax

CT scan revealed an aberrant subclavian artery (white arrow), giving the

impression of a mediastinal mass

CASE

A 79 -year-old female patient applied to clinics other

than ours with complaints of dysphagia and cough for

around 1 year. She had been treated for pneumonia and

with antireflux treatments, and was suspected of having a

mediastinal mass based on a non-contrast thorax CT,

leading to her referral to our center.

Upon referral, no pathology was detected during a physical

examination and chest X-ray (Figure 1). The patient

reported choking and swallowing difficulties, especially

while eating solid foods, for the last year. She had suffered

from an MCA (mean cerebral artery) infarction

about 30 years earlier. Her left ventricular ejection fraction

was 30–35%, and she had a pacemaker. No abnormal

findings were detected in biochemical tests. A

barium esophagogram revealed esophageal compression

(Figure 2).

An attempted endoscopy failed due to an obstruction of

the esophageal lumen. A contrast-enhanced thorax CT

and neck CT revealed that the patient’s right subclavian

artery emerged from the distal arch of the aorta and

coursed posterior to the esophagus at the level of T2-T3

vertebrae. The patient was subsequently diagnosed with

aberrant subclavian artery. CT images of the ARSA revealed

esophageal compression, but without any tracheal

compression (Figure 3). The diagnosis was confirmed

from sagittal and coronal CT sections (Figure 4).

Surgical treatment has not been considered in the patient

who underwent cardiovascular surgery consultation for

the surgical treatment of ARSA, due to advanced age of

the patients and the absence of symptoms. Consuming

soft foods and avoiding foods and beverages that could

affect esophageal motility have been recommended.

Figure 2: Stenosis (white arrow) due to the compression of the aberrant

subclavian artery on a barium X-ray. The aspiration of the contrast

material into the bronchial system and the patient's pacemaker can also

be seen

DISCUSSION

In cases with ARSA anomalies, four arteries arise from the

aortic arch – the right main carotid artery, the left main

carotid artery, the left subclavian artery and the ARSA

(Figure 5). As the final branch, the ARSA, arises proximal

to the descending aorta in the left hemithorax, and then

courses up, passing around the back of the esophagus.

This is the most common variation, with a prevalence of

80–84% (3). Other variations pass between the esophagus

and trachea in 12.7–15% and in front of the trachea

in 4.2–5% of cases (4)

When ARSA, which is often asymptomatic and detected

incidentally, becomes symptomatic, patients may present

with complaints of difficulty swallowing, regurgitation,

postprandial bloating, chest pain and cough, especially

when eating solid foods. In a more recent study, however,

dysphagia was reported as a symptom in 71.2% of 141

ARSA cases (5).


Incidentally Detected Aberrant Right Subclavian Artery: A Case Report | Ağar et al.

Figure 3: Aberrant right subclavian artery (yellow arrow) detected in

contrast-enhanced thorax CT and the esophagus constricted by the

pressure of the artery (blue arrow)

Figure 4: The aberrant right subclavian artery (yellow arrow) and compressed

esophagus (blue arrow) on sagittal (top) and coronal (bottom)

CT scans of the patient

allowing the vascular structures and surrounding tissues

to be examined as a whole (7).

Diagnoses of ARSA can also be made based on the detection

of compression on esophagograms. In the esophagoscopy

diagnostic approach, pulsations on the posterior

wall of the esophagus are the most common finding,

and transesophageal echocardiography may also aid in

the diagnosis (8).

Dietary and pharmacological therapies are often the first

treatment options in ARSA, while surgical treatments are

recommended for patients who do not respond to medical

treatment, or whose complaints aggravate. Available

surgical treatments include dissection of the aberrant

artery and its anastomosis up to the ipsilateral carotid

artery, although due to the high mortality rates reported

in surgical treatments, endovascular treatment methods

have been recommended more recently (9). Generally

performed surgeries include posterior mediastinal procedures

or occlusion of the ARSA with a right pleural approach

in a median sternotomy, followed by an extraanatomical

axillary artery bypass and several reconstructive

procedures (10,11).

CONCLUSION

ARSA should be considered in the differential diagnosis of

patients who present with dysphagia with or without

chronic cough. Imaging methods such as contrastenhanced

thorax CT, barium esophagogram and

transesophageal echocardiography can aid in the diagnosis.

While conservative treatments are preferred in

cases of ARSA with asymptomatic or mild symptoms,

surgical treatment options may be preferred in severe

cases.

Figure 5: Schematic view of aberrant right subclavian artery anomaly

ARSA is usually diagnosed in middle and advanced ages,

and therefore clinical findings are delayed. Anatomical

and physiological changes, such as increases in esophageal

and arterial wall rigidity due to atherosclerosis, and

aortic elongation with aging have been suggested to lead

to delays in the identification of clinical findings (6).

Although angiography has traditionally been the optimum

diagnostic method of ARSA, 3-dimensional spiral CT

angiography is accepted as the basic imaging method for

the diagnosis of thoracic vascular anomalies. Contrastenhanced

CT scans, which are widely used, are superior

to conventional angiography, being noninvasive and


None declared.


Concept - M.A., S.K.; Planning and Design - M.A., S.K.;

Supervision - M.A., S.K.; Funding - M.A., S.K.; Materials -

M.A.; Data Collection and/or Processing - M.A., S.K.;

Analysis and/or Interpretation - M.A., S.K.; Literature

Review - M.A.; Writing - M.A., S.K.; Critical Review -

M.A., S.K.

YAZAR KATKILARI

Fikir - M.A., S.K.; Tasarım ve Dizayn - M.A., S.K.; Denetleme

- M.A., S.K.; Kaynaklar - M.A., S.K.; Malzemeler -

M.A.; Veri Toplama ve/veya İşleme - M.A., S.K.; Analiz



ve/veya Yorum - M.A., S.K.; Literatür Taraması - M.A.;

Yazıyı Yazan - M.A., S.K.; Eleştirel İnceleme - M.A., S.K.

REFERENCES

1. Bayford D. An account a singular case of obstructed

deglutition. Memoirs Med Soc London 1794; 2: 275-86.

2. Polednak AP. Prevalence of the aberrant right subclavian

artery reported in a published systematic review of cadaveric

studies: the impact of an outlier. Clin Anat 2017;

30:1024-8. [CrossRef]

3. Zingarelli A, Morelli MC, Seitun S, Bezante GP, Balbi M,

Brunelli C. Aberrant right subclavian artery (arteria lusoria)

challenging 4-French homolateral transradial coronary

catheterization in adulthood. Heart Lung Circ

2015; 24:e164-8. [CrossRef]

4. Holzapfel G. Ungewöhnlicher Urpsrung und Verlauf der

Arteria subclavia dextra. Anat Hefte 1899; 12:369-523.

[CrossRef]

5. Polguj M, Chrzanowski Ł, Kasprzak JD, Stefańczyk L, Topol

M, Majos A. The aberrant right subclavian artery (arteria

lusoria): The morphological and clinical aspects of

one of the most important variations-a systematic study of

141 reports. ScientificWorldJournal 2014; 2014:292734.

[CrossRef]

6. Janssen M, Baggen MG, Veen HF, Smout AJ, Bekkers JA,

Jonkman JG, et al. Dysphagia lusoria: clinical aspects,

manometric findings, diagnosis, and therapy. Am J Gastroenterol

2000; 95:1411-6. [CrossRef]

7. Türkvatan A, Büyükbayraktar FG, Olçer T, Cumhur T.

Multidetector computed tomographic angiography of aberrant

subclavian arteries. Vasc Med 2009; 14: 5-11.

[CrossRef]

8. Deck M, Grocott HP, Yamashita MH. Aberrant right subclavian

artery: an impediment to transesophageal echocardiography.

Can J Anaesth 2021; 68: 423-4.

[CrossRef]

9. Attmann T, Brandt M, Müller-Hülsbeck S, Cremer J. Twostage

surgical and endovascular treatment of an aneurysmal

aberrant right subclavian (Lusoria) artery. Eur J

Cardiothorac Surg 2005; 27:1125-7. [CrossRef]

10. Ikeno Y, Koda Y, Yokawa K, Gotake Y, Henmi S, Nakai

H, et al. Graft replacement of Kommerell diverticulum

and in situ aberrant subclavian artery reconstruction. Ann

Thorac Surg 2019; 107:770-9. [CrossRef]

11. Kurisu K, Imasaka KI, Hashino A, Ueno Y, Shiose A.

Pleural approach to aberrant right subclavian artery in

aortic surgery. Ann Vasc Dis 2021; 14:249-51.

[CrossRef]



OLGU SUNUMU

CASE REPORT

A Case of Suspected COVID-19 Identified with

AIDS, PCP and Tuberculosis

AIDS, PCP ve Tüberküloz Tanıları Alan COVİD-19 Şüpheli bir Olgu

Zeynep Tilbe Saymaz,

Şeref Özkara


Abstract

HIV (Human Immunodeficiency Virus) is the virus that

causes AIDS (Acquired Immune Deficiency Syndrome),

while PCP (Pneumocystis jiroveci pneumonia), tuberculosis,

CMV (Cytomegalovirus) and candidiasis are

the OIs (opportunistic infections) occurring due to

immune deficiency. OIs, and bacterial pneumonias in

particular, are the most common causes of mortality,

which makes the screening and prophylactic therapy

for OIs necessary. The synergy between tuberculosis

and HIV has long been known, and worsens the

prognosis. PCP is an OI that is caused by a fungus

named P. jiroveci. COVID-19 has emerged as a new

cause of death among AIDS patients. Screening and

prophylactic therapy for Ols is vital for patients with

AIDS, however, mortality may be high due to delays

in screening and prophylaxis in those whose HIV

positivity is detected coincidentally. We report here on

a patient who applied to our hospital with suspected

COVID-19 pneumonia who was found during followup

to be HIV positive with PCP and pulmonary tuberculosis.

Key words: HIV, PCP, Tuberculosis, Covid-19.

Öz

HIV (İnsan İmmün Yetmezlik Virüsü) immün sistemi

zayıflatan, AIDS (Kazanılmış Bağışıklık Yetersizliği

Sendromu) hastalığına yol açan viral bir enfeksiyondur.

İmmün sistemin zayıflaması ile birlikte, tüberküloz,

PCP (Pneumocystis jiroveci pnömonisi), CMV

(sitomegalovirüs), kandida gibi fırsatçı enfeksiyonlara

olanak sağlamaktadır. En yüksek mortaliteyi oluşturan

durumlar fırsatçı enfeksiyonlardır. Bu durum AIDS

hastalarında fırsatçı enfeksiyonlara yönelik tarama ve

profilaksi uygulanmasını gerekli kılmıştır. Tüberküloz

ile HIV birlikteliği uzun zamandır bilinmekte ve prognozu

kötüleştirmektedirler. PCP ise günümüzde mantar

olarak kabul edilen P. jirovecii etkenli fırsatçı bir

pnömonidir. Covid -19 pandemisi ile birlikte de AIDS

hastalarında yeni bir mortalite sebebi ortaya çıkmıştır.

AIDS hastalarının bu fırsatçı enfeksiyonlara karşı

taranması, profilaktik tedavi alması ve erkenden

tedavi almaları hayati önem taşımaktadır. Ancak HIV

pozitifliği rastlantısal olarak saptanan kişilerde tarama

ve profilaksi gecikmesinden ötürü mortalite yüksek

seyredebilmektedir. Bu yazıda Covid-19 pnömonisi

şüphesi ile hastanemize başvuran bir hastanın HIV

pozitif tespit edilip PCP ve takibinde akciğer tüberkülozu

saptanması anlatılmaktadır.

Anahtar Sözcükler: HIV, PCP, Tüberküloz, Covid-19.

Ankara Atatürk Respiratory Disease and Thoracic Surgery

Hospital, Ankara, Türkiye

Ankara Atatürk Gçğüs Hastalıkları ve Göğüs Cerrahisi Eğitim

ve Araştırma Hastanesi, Ankara


Correspondence (İletişim): Zeynep Tilbe Saymaz, Ankara Atatürk Respiratory Disease and Thoracic Surgery Hospital, Ankara, Türkiye

e-mail: tilbesaymaz@gmail.com

102


Human immunodeficiency virus (HIV) is a viral infectious

disease that weakens the immune system and affects

blood cells, especially CD4-T lymphocytes (1). HIV remains

as a growing health problem worldwide. According

to the World Health Organization (WHO), 38 million

people worldwide were infected with HIV in 2019, while a

total of 75 million people have died of acquired immune

deficiency syndrome (AIDS) since the epidemic began (1).

Due to the immunodeficiency associated with AIDS, opportunistic

infections and some cancers are common,

including tuberculosis (TB) and Pneumocystis jirovecii

pneumonia (PCP). Accordingly, WHO suggests screening

for TB and other opportunistic infections in patients with

HIV (1,2). There is a synergistic relationship between TB

and HIV positivity (3,4).

PCP is the most common infectious disease in AIDS.

Pneumocystis jirovecii is the microbiological fungal pathogen

that causes PCP (5).

Mortality in AIDS has decreased as a result of screening

tests for infections and antiretroviral treatments (ART) (5,6).

Today, only those who are unaware of their HIV positivity

can contract such opportunistic infections (6).

In December 2019, a SARS Cov-2 virus outbreak in Wuhan,

China led to a pandemic that came to affect the

world, and the resulting COVID-19 disease has become

the highest cause of mortality in AIDS patients. The clinical

findings and radiological images of COVID-19 can

be mistaken for opportunistic infections in HIV-positive

patients, leading to a new problem in the form of treatment

delays (7).

Our case presented with the clinical and radiological

suspicion of COVID-19 pneumonia, but was subsequently

diagnosed with AIDS and PCP. During treatment for

ART and PCP, the patient was also found to have TB.

middle lobe of the right lung with a diffusely groundglassed

bilateral appearance (Figure 2 and 3). Interstitial

lung disease and COVID-19 pneumonia could not be

differentiated in the city hospital, and so the patient was

referred to us.

Upon physical examination, the patient's general condition

was good, blood pressure was 110/70 mmHg, heart

rate was 108 beats/min, respiratory rate was 20, fever

was 38.3˚C, oxygen saturation in room air was 97% and

respiratory sounds were decreased bilaterally.

Laboratory tests revealed hemoglobin 13.8 g/dl, creatinine

1.29 mg/dl, GFR 60.3%, serum lactate dehydrogenase

(LDH) 453 IU/L, C-reactive protein 74 mg/L,

ferritin 1612 ng/ml and d-dimer 0.88 mg/L.

Figure 1: Chest X-ray of the patient at the time of admission

CASE

Our 63-year-old male patient was born in Bartın, Turkey,

and was employed as a construction worker. He had no

chronic disease and did not use drugs, but had a 40-

pack years smoking history.

He presented at Ankara City Hospital with complaints of

shortness of breath, cough, sputum for 2 months and

diarrhea for 2 weeks. And weight loss of 5 kg in 2 months.

A radiograph of the posterior-anterior chest revealed

infiltration extending from the vertebrae to the periphery

under the right hilar region (Figure 1). Thoracic computed

tomography (CT) revealed a millimetric nodule in the

Figure 2: A patient thorax tomography revealing the dark bronchus sign

image that is characteristic of PCP


A Case of Suspected COVID-19 Identified with AIDS, PCP and Tuberculosis |Saymaz et al.

The patient was admitted to the hospital with suspected

COVID-19 disease, however two COVID-19 polymerase

chain reaction (PCR) tests were both negative. The patient

was treated with favipiravir, enoxaparin sodium, ipratropium

bromide, salbutamol and ceftriaxone. After an anti-

HIV test result came back positive, the patient was consulted

with the infectious diseases doctor. The patient was

considered to have PCP, and ceftriaxone was switched to

trimethoprim-sulfamethoxazole (TMP-SMX) treatment.

After the PCP PCR test was positive, the patient was referred

to the infectious disease service.

Upon follow-up, the patient’s CD4 count was 135/mm3

and PCP direct fluorescent antibody was positive, and

treatment was started with emtricitabine, tenofovir alafenamide

and bictegravir, in addition to treatment with TMP-

SMX. During follow-up, the patient's oxygen saturation at

room air declined to 66%, fever was 38.5oC, and blood

cultures were all negative. The patient, who was unable

to provide a sputum sample, was referred to us again for

fiberoptic bronchoscopy (FOB), bronchial lavage acidfast

staining (AFS) and bronchial lavage Xpert MTB/RIF

(rapid PCR test). The patient's bronchial lavage AFS

smear was negative, while the Xpert MTB/RIF test detected

Mycobacterium tuberculosis (MTB) and rifampicin was

susceptible. Fifteen colonies were grown in the culture,

and MTB were detected that were sensitive to Isoniazid

(H), Rifampicin (R), Streptomycin (S), Pyrazinamide (Z) and

Ethambutol (E) on the antibiogram. The patient was diagnosed

with HIV+, Xpert MTB/RIF positive new case of

pulmonary tuberculosis and treated with H, R, E and Z.

The patient's fever did not return after TB treatment. Nausea

and vomiting were found in relation to the isoniazid

intake, and gastrointestinal intolerance of the drug was

diagnosed, and so treatment with R, E, Z, and moxifloxacin

was started.

A post-TB diagnosis CT showed bilateral diffuse micrographs

that had increased significantly when compared to

the previous CT, indicating PCP (Figure 4).

Figure 3: Dark bronchus sign and bilateral diffuse ground-glass densities

in the patient thorax tomography

Figure 4: Imaging of the patient after diagnosis with tuberculosis

DISCUSSION

Since the sensitivity of COVID-19 PCR tests in the early

phase of the disease is 71%, patient was started on favipiravir

treatment for COVID-19 pneumonia based on

their complaints on admission and the ground-glass images

on CT, even though the COVID-19 PCR test was

negative (8). The ground-glass images on the patient's CT,

however, were not typical of COVID-19 pneumonia, and

the patient's symptoms and radiological imaging were

actually attributable to the patient’s diagnosis of HIV and

PCP during hospitalization. It was assumed that the patient

did not have COVID-19 pneumonia.

With the advances in HIV treatment, mortality from the

disease has declined, although as opportunistic infections

occurring alongside HIV infection can lead to death,

screening, prophylaxis, early diagnosis and treatment

must be carried out thoroughly in HIV patients (1).

It is well known that those with HIV-infections are at high

risk of developing TB, and that a synergistic relationship

exists between the two (3,9). DeRiemer et al. (10) reported

that the probability of developing pulmonary TB in

HIV-positive individuals is 8 times higher than in non-HIVpositive

individuals. To date, more than 40 million people

have lost their lives due to the comorbidity of HIV and TB,

which necessitates screening and prophylaxis for TB in

those who are HIV positive. The tuberculin skin test (TST)

and interferon-gamma release assays are used for the

screening of tuberculosis in HIV-positive patients. It is

important to remember that HIV-infected patients must be

considered immunosuppressed when interpreting TST

results, and so results of 5 mm and above should be

considered positive. In Turkey isoniazid is given for 9

months as TB prophylaxis as the first choice. Imaging of

the lungs in immunocompromised patients, such as those

who are HIV-positive, may be normal, or there may be

such atypical findings as pleural fluid, pneumothorax,

mass lesions and miliary shadows. Our patient did not

have typical imaging for TB. AFS, Xpert MTB/RIF or the

TB culture of specimens can be used for diagnosis. The

bacteriological positivity rate is lower in HIV positive TB



patients than in immune competent TB cases. In recent

years, a serological urine test has been developed for

lipoarabinomannan for TB diagnosis in HIV-positive patients.

The duration of TB treatment in HIV-positive patients

is 6 months (9). Mortality in multidrug-resistant TB

patients is increased in HIV patients (3). Drug susceptibility

testing should be performed as soon as possible and

appropriate treatment should be started early. Nontuberculous

Mycobacteria (NTM) infection is also more

common in HIV-infected patients than in non-HIV patients,

although MTB is more common than NTM in HIV patients.

Some HIV-TB patients who take their medications regularly

and respond well to treatment may experience radiographic

and clinical deterioration, which is a condition

referred to as immune reconstitution inflammatory syndrome

(IRIS) that is associated with the recovery of immunity.

Our patient did not develop IRIS. Antiretroviral

treatment and tuberculosis treatment should be continued

together without interruption (9). Studies have shown

mortality to be lower in patients treated with ART in the

early period after starting anti TB treatment (9,11), although

ART may interact with anti-TB drugs, especially

rifampicin, which may be replaced with rifabutin.

PCP is an opportunistic infectious disease that occurs in

immunosuppressed patients, and can be mortal in HIV

positive patients in whom it develops when the CD4 T

lymphocyte count falls below 200/mm3. In our case, the

CD4 count was 135/mm3. Patients may develop nonspecific

symptoms such as fever, cough, sputum and

shortness of breath (5,6), while our patient had shortness

of breath, cough, fever, and diarrhea. As the patient was

examined during the COVID-19 pandemic, COVID-19

pneumonia was initially thought to be present. Imaging of

the patient's lungs may be normal, although pneumothorax,

especially in HIV-positive individuals, should be suggestive

of PCP (6). P. jirovecii does not grow in cultures. A

definitive diagnosis can be confirmed based on morphological

evidence of the microorganism in bronchoalveolar

lavage (BAL), with sputum collected by FOB. Immunofluorescence

staining with fluorescently labeled monoclonal

antibodies is considered the optimum means of diagnosis.

In some studies, an LDH level of >500 IU/L has been

shown to have a poor prognosis in PCP (6). Our patient’s

LDH level was 453 IU/L. TMP-SMX is recommended as a

prophylaxis and for treatment of pneumonia in HIVpositive

patients with PCP. Dose adjustment is important

in treatment, with TMP 15-20 mg–SMX 75-100

mg/kg/day IV being given in 3–4 doses. It has been reported

that treatment with corticosteroids in patients with

hypoxia has a positive effect on prognosis (6). While

receiving TMP-SMX treatment for PCP, outpatient also

received corticosteroid treatment due to hypoxia, and

emtricitabine, tenofovir, alafenamide and bictegravir as

antiretroviral treatments.

In a study by Castro et al. (12), the coexistence of PCP

and TB was observed in 5.8% of a total of 2,651 HIV

positive patients, while a study by Tchatchouang et al. (13)

revealed the coexistence of PCP and TB in 10% of a total

of 211 HIV positive patients. TB shouldn’t be forgotten,

as radiology may not be typical or may even be normal in

HIV positive patients. Although radiology is typical for

PCP, as in our patient, TB treatment was initiated as a

result of the patient’s positive TB PCR and positive culture.

In summary, a patient who was thought to have COVID-

19 pneumonia was diagnosed with HIV positivity, PCP

and TB, and was treated successfully.


None declared.


Concept - Z.T.S., Ş.Ö.; Planning and Design - Z.T.S.,

Ş.Ö.; Supervision - Z.T.S., Ş.Ö.; Funding - Z.T.S.; Materials

- Z.T.S.; Data Collection and/or Processing - Z.T.S.;

Analysis and/or Interpretation - Z.T.S.; Literature Review -

Z.T.S.; Writing - Z.T.S.; Critical Review - Z.T.S.

YAZAR KATKILARI

Fikir - Z.T.S., Ş.Ö.; Tasarım ve Dizayn - Z.T.S., Ş.Ö.;

Denetleme - Z.T.S., Ş.Ö.; Kaynaklar - Z.T.S.; Malzemeler

- Z.T.S.; Veri Toplama ve/veya İşleme - Z.T.S.; Analiz

ve/veya Yorum - Z.T.S.; Literatür Taraması - Z.T.S.; Yazıyı

Yazan - Z.T.S.; Eleştirel İnceleme - Z.T.S.

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4. Chepkondol GK, Jolly PE, Yatich N, Mbowe O, Jaoko

WG. Types and prevalence of HIV-related opportunistic

infections/conditions among HIV-positive patients atten-


A Case of Suspected COVID-19 Identified with AIDS, PCP and Tuberculosis |Saymaz et al.

ding Kenyatta National Hospital in Nairobi, Kenya. Afr

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R, Tugiran M, et al. Laboratory findings and clinical

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infection among HIV-infected patients with pulmonary

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tesadüfen tanı konulan AIDS olgusu. Flora Dergisi 2019;


7. Blanco JL, Ambrosioni J, Garcia F, Martínez E, Soriano A,

Mallolas J, et al. COVID-19 in patients with HIV: clinical

case series. The Lancet HIV 2020; 7:e314-6. [CrossRef]

8. Ceylan N, Savaş R. Radiological findings of COVID-19

pneumonia. Eurasian J Pulmonol 2020; 22:19-24.

[CrossRef]

9. Türkiye Cumhuriyeti Sağlık Bakanlığı. Tüberküloz tanı ve

tedavi kılavuzu. (Republic of Turkey, Ministry of Health.

Tuberculosis Diagnosis and Treatment Guidelines). Ankara

2019.

10. DeRiemer K, Kawamura LM, Hopewell PC, Daley CL.

Quantitative impact of human immunodeficiency virus infection

on tuberculosis dynamics. Am J Respir Crit Care

Med 2007; 176:936-44. [CrossRef]

11. Abdool Karim S S, Naidoo K, Grobler A, et al. Integration

of antiretroviral therapy with tuberculosis treatment.

N Engl J Med 2011; 365: 1492-501. [CrossRef]

12. Castro JG, Manzi G, Espinoza L, Campos M, Boulanger

C. Concurrent PCP and TB pneumonia in HIV infected

patients. Scand J Infect Dis 2007; 39:1054-8. [CrossRef]

13. Tchatchouang S, Nzouankeu A, Donkeng V, Eyangoh S,

Ngando L, Penlap V, et al. Prevalence of opportunistic

pathogens Pneumocystis jiroveci and tubercle bacilli in

HIV-infected patients with respiratory infections in Yaounde,

Cameroon. AIDS Res Hum Retroviruses 2019;




OLGU SUNUMU

CASE REPORT

Young Female COVID-19 Patient Presenting

with Epileptic Seizure

Epileptik Nöbetle Başvuran Genç Kadın COVID-19 Hastası

Sümeyye Kement 1 , Kader Topçu 1 , Cem Arda Yacan 1 , Seda Berke 1 , Nilgün Cengiz 2 ,

Oğuz Uzun 1


Abstract

COVID-19 is a new type of coronavirus infection with

a wide clinical spectrum, ranging from asymptomatic

to severely symptomatic, and that mostly affects the

respiratory tract. Although the respiratory tract is the

primary area affected by the disease, neurological

symptoms such as headache, dizziness and muscle

ache have also been reported in some patients since

the early stages of the pandemic. COVID-19 symptoms

and complications can affect the peripheral and

central nervous systems as well as the skeletal muscles,

while epileptic seizure is a rare manifestation of

COVID-19. We present here the case of a female

patient admitted to hospital with epileptic seizure due

to COVID-19 encephalopathy.

Key words: COVID-19, seizures, encephalopathy.

Öz

COVID-19, asemptomatikten şiddetli semptomlarla

giden hastalığa kadar geniş bir klinik spektrum gösteren,

çoğunlukla solunum yollarını etkileyen yeni tip

koronavirüs enfeksiyonudur. Hastalıkta temel etkilenen

bölge solunum sistemi olsa da, pandeminin

erken evrelerinden itibaren bazı hastalarda, baş ağrısı,

baş dönmesi ve kas ağrıları gibi nörolojik semptomlar

da bildirilmiştir. COVID-19 semptomları ve

komplikasyonları, hem periferik hem de santral sinir

sistemini ve iskelet kaslarını etkileyebilir. Epileptik

nöbet, COVID-19'un nadir bir belirtisidir. Biz de

COVID-19 ensefalopatisi nedeniyle, hastaneye epileptik

nöbet ile başvuran bir kadın hastayı sunuyoruz.

Anahtar Sözcükler: COVID-19, nöbet, ensefalopati.

1 Department of Pulmonary Medicine, Ondokuz Mayıs University,

Samsun, Türkiye

2 Department of Neurology, Ondokuz Mayıs University, Samsun,

Türkiye

1 Ondokuz Mayıs Üniversitesi, Göğüs Hastalıkları Anabilim

Dalı, Samsun

2 Ondokuz Mayıs Üniversitesi, Nöroloji Anabilim Dalı, Samsun


Correspondence (İletişim): Sümeyye Kement, Department of Pulmonary Medicine, Ondokuz Mayıs University, Samsun, Türkiye

e-mail: sumeyyeekement@gmail.com

107


Coronavirus disease 2019 (COVID-19) is a new type of

coronavirus infection that was first defined as an atypical

pneumonia epidemic in the city of Wuhan, China in December

2019, and was later upgraded to a pandemic by

the World Health Organization (WHO) on March 11,

2020. COVID-19 presents with a wide clinical spectrum

that ranges from asymptomatic to severely symptomatic,

and that mostly affects the respiratory tract (1). COVID-

19 is caused by the recently identified severe acute respiratory

distress syndrome coronavirus 2 (SARS-CoV-2),

and is an ongoing global health emergency (2).

We present here a case who applied to the hospital

emergency service with seizure that was treated as meningitis,

but was later evaluated to be encephalopathy

related to COVID-19.

CASE

A 19-year-old female patient with no known disease

referred to the emergency service with complaints of fever,

fatigue and headache that did not respond to the antipyretics

she used at home. An oropharyngeal swab for

COVID-19 was subjected to a real-time Reverse Transcription-Polymerase

Chain Reaction (RT-PCR) test and

the patients symptoms were treated, including metpamide,

and she was discharged. After experiencing dizziness at

03:00 on the same day, her speech gradually deteriorated

and she became incomprehensible, and meaningless

speech, empty gaze and disorientation developed. Tremors

were noted in the right arm and leg, along with

spasms in the left arm and leg, and balance disorder

developed. She reapplied to the emergency department

with these complaints, reporting no sleep disturbance,

long-term hunger or head trauma before the seizure. The

patient produced loud moans, meaningless sounds and

empty gazes, while system examinations were normal.

The patient's serum creatinine was 0.54 mg/dl, C-

reactive protein (CRP) 3.3 mg/l (reference range: 0-5),

white blood cell count 5.9 109/L, lymphocyte 0.7 109/L

(12%), platelet 241 109/L, hemoglobin 14.3 g/dl and d-

dimer 1,33 mg/L (reference range: 0-0.55). No abnormal

findings were detected on chest X-ray or thorax computed

tomography (Figure 1), and the seizure was thus

evaluated as an extrapyramidal side effect of metpamide

use. The patient was followed up in the general intensive

care unit due to the continued confusion and contractions

of extremities. A nasopharyneal RT-PCR test for SARS-

CoV-2 was positive and brain magnetic resonance imaging

(MRI) produced normal findings. A lumbar puncture

analysis revealed a protein level of 50.7 mg/dl in cerebrospinal

fluid (CSF). The CSF cell count was 14µL-10

mononuclear and 2 polymorphonuclear cells without red

blood cells. Due to the fever, the high CRP of 68 mg/L

and the white blood cell count of 11.4 109/L, the patient

was started on antiepileptic, favipravir, ceftriaxone,

levofloxacin and vancomycin treatments with an initial

diagnosis of meningitis and COVID-19.

On the sixth day of hospitalization the patient was transferred

to the inpatient service after her state of consciousness

improved. She was able to make short sentences

and to respond to single commands. On the second day

of inpatient service follow-up, her seizures recurred, and

she was returned to the intensive care unit as her oxygen

saturation decreased to 88%. A further lumbar puncture

was made on the ninth day of hospitalization: the CSF

protein (15 mg/dl) and glucose (75 mg/dl) values were

within the normal range; mycobacteria PCR and ARB

(Acid Resistant Bacilli) in the CSF were negative. No mycobacteria

or any other microorganisms were cultured

from CSF. Brucella Coombs and Rose Bengal agglutination

tests in the CSF were negative, and so the diagnosis

of meningitis was excluded. The patient was in a state of

confusion, but her comprehension and word output was

intact. As lobar consolidation observed in the PA chest X-

ray, taken on the 10th day of hospitalization, meropenem

antibiotherapy was given for 14 days with a diagnosis of

hospital-acquired pneumonia (Figure 2). On the 11th day

of hospitalization, the patient was taken to the inpatient

service for further follow-up as her general condition was

improving and there had been no recurrence of seizures.

Electroencephalography (EEG) taken on the 15th day was

consistent with moderate encephalopathy (Figure 3a),

and brain MRI taken on 19th day was normal. In the light

of the patient’s clinical, laboratory and radiological findings,

her condition was evaluated as encephalopathy

secondary to COVID-19. Levetiracetam was given from

the beginning of admission to the hospital, and oxygen

support with nasal cannula was given during hospitalization.

The patient was discharged on the 23rd day of hospitalization

due to the regression of infiltrations on chest

X-ray, the improvement of oxygen saturation, the regression

of acute phase reactants and the completion of antibiotic

therapy (Figure 4). At discharge, CRP was <3 mg/l

and white blood cell count was 7.82 109/L. It was

planned to proceed with levetiracetam treatment for 6

months due to the patient’s history of seizures.

When the patient applied for follow-up control examination

25 days after discharge, her complaints were dimin-


Young Female COVID-19 Patient Presenting with Epileptic Seizure | Kement et al.

ished. The control EEG was consistent with mild encephalopathy

(Figure 3b).

DISCUSSION

COVID-19 is a pandemic disease that presents with clinical

pictures ranging from mild illness with non-specific

symptoms to respiratory failure with acute respiratory

symptoms, and to severe pneumonia and sepsis (3). Epileptic

seizure is a rare manifestation of COVID-19 (4).

Coughs, fever, fatigue and shortness of breath are common

symptoms of COVID-19, and many neurological

abnormalities have been reported in patients with

COVID-19 from the beginning of the pandemic. Among

these, loss of smell and taste are distinctive symptoms that

were later added to the clinical findings of the disease.

The respiratory tract is the primary affected area in the

disease, although neurological symptoms such as headache,

dizziness and muscle ache have also been reported

since the early stages of the pandemic. Neurological

involvement in COVID-19 is a considerable component

of the disease, as it has been reported that approximately

90% of patients with a diagnosis of COVID-19 have at

least one subjective neurologic complaint (3). COVID-19

symptoms and complications can affect the peripheral

and central nervous systems as well as skeletal muscles

(2).

Figure 1: Chest X-ray upon presentation with no abnormal finding

Figure 2: Lobar consolidation in the right lower zone of the lung

Viral neuroinvasion may occur via several routes, including

transsynaptic transfer across infected neurons, entry

via the olfactory nerve, infection of vascular endothelium

or leukocyte migration across the blood-brain barrier (5).

Neurological involvement in COVID-19 can present with

many neurological manifestations, such as encephalopathy,

encephalitis, ischemic stroke and postinfectious neurological

complications (6).

Neurological manifestations in SARS-CoV-2 infection due

to encephalopathy/encephalitis and acute cerebrovascular

disease have been observed in up to 8% of patients

with severe disease (7). There have been a few cases of

COVID-19 fulfilling the diagnostic criteria for infectious

encephalitis (8,9), with the main findings in such cases

being altered mental status, fever, seizures, white blood

cells in CSF and focal brain abnormalities on neuroimaging.

SARS-CoV-2 has been detected in the CSF in two

patients (10,11), and temporal lobe encephalitis was

confirmed by biopsy that showed perivascular lymphocytic

infiltrates and hypoxic neuronal damage in one patient

(8).

In the study by Meppiel et al. (12), 222 COVID-19 cases

from 46 centers in France with neurological involvement

were reviewed. COVID-19- associated encephalopathy

was observed most commonly in 30% of the patients,

followed by acute ischemic cerebrovascular disease in

25%, encephalitis in 9.5% and Guillian Barre Syndrome

in 6.8%.



Figure 4 The regression of infiltrations on chest X-ray

Figure 3a and b: Initial EEG showing delta and theta waves compatible

with moderate encephalopathy (a), and follow-up EEG with theta waves

compatible with mild encephalopathy (b)

COVID-19 patients may initially refer to clinics with

headache, fever and new-onset seizure. In some COVID-

19 patients, SARS-CoV-2 was found in the CSF, showing

that this neurological manifestation can be attributed to

the virus. However, COVID-19 patients presenting with

acute meningoencephalitis with neither SARS-CoV-2 nor

other viral pathogens detected in the CSF have also been

seen. As such, SARS-CoV-2- RNA undetected in the CSF

may indicate that direct brain infection is not the only

means of neuroinvasion, and that other possible ways of

transmission may exist, such as peri-infectious inflammation

and altered neurotransmission, as underlying reasons

for meningoencephalitis (2). Lymphocytic pleocytosis may

be seen in the CSF of COVID-19 patients (13,14). Since

meningoencephalitis can be complicated by intracerebral

and subdural hematomas, early detection of the virus is

essential to ensure the start of appropriate treatment and

to prevent the onset of hemorrhagic encephalopathy,

which can lead to severe disability or life-threatening

situations (2).

Such neurological involvements as encephalopathy, cerebrovascular

disease and Guilian Barre Syndrome can

also complicate in the course of COVID-19 infection (1).

Our patient presented with prominent neurological symptoms

rather than respiratory symptoms, suggesting that

COVID-19 infection should be kept in mind in patients

presenting with epileptic seizure. Encephalopathy and

encephalitis may develop during the hospitalization of

COVID-19 patients who present initially with respiratory

symptoms (15).

CONCLUSION

Healthcare professionals should be aware that COVID-

19 patients may develop encephalopathy at admission or

during hospitalization, and so all appropriate examinations

and investigations should be performed.


None declared.


Concept - O.U., S.K., K.T., C.A.Y., S.B., N.C.; Planning

and Design - S.K., O.U., K.T., C.A.Y., S.B., N.C.; Supervision

- O.U., S.K., K.T., C.A.Y., S.B., N.C.; Funding -

C.A.Y., S.B., N.C.; Materials - C.A.Y., S.B., N.C.; Data

Collection and/or Processing - S.B., C.A.Y., N.C.; Analysis

and/or Interpretation - K.T., S.K., O.U.; Literature

Review - O.U., S.K.; Writing - K.T., S.K., O.U.; Critical

Review - K.T., O.U., S.K.


Young Female COVID-19 Patient Presenting with Epileptic Seizure | Kement et al.

YAZAR KATKILARI

Fikir - O.U., S.K., K.T., C.A.Y., S.B., N.C.; Tasarım ve

Dizayn - S.K., O.U., K.T., C.A.Y., S.B., N.C.; Denetleme

- O.U., S.K., K.T., C.A.Y., S.B., N.C.; Kaynaklar - C.A.Y.,

S.B., N.C.; Malzemeler - C.A.Y., S.B., N.C.; Veri Toplama

ve/veya İşleme - S.B., C.A.Y., N.C.; Analiz ve/veya

Yorum - K.T., S.K., O.U.; Literatür Taraması - O.U., S.K.;

Yazıyı Yazan - K.T., S.K., O.U.; Eleştirel İnceleme - K.T.,

O.U., S.K.

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F, Ghasemi M. SARS-CoV-2 and nervous system:

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2. Harapan BN, Yoo HJ. Neurological symptoms, manifestations,

and complications associated with severe acute

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coronavirus disease 19 (COVID-19). J Neurol 2021;

268:3059-71. [CrossRef]

3. Liguori C, Pierantozzi M, Spanetta M, Sarmati L, Cesta N,

Iannetta M, et al. Subjective neurological symptoms

frequently occur in patients with SARS-CoV2 infection.

Brain Behav Immun 2020; 88:11-6. [CrossRef]

4. Keshavarzi A, Janbabaei G, Kheyrati L, Ghavamabad LH,

Asadi-Pooya AA. Seizure is a rare presenting manifestation

of COVID-19. Seizure 2021; 86:16-8. [CrossRef]

5. Zubair AS, McAlpine LS, Gardin T, Farhadian S, Kuruvilla

DE, Spudich S. Neuropathogenesis and neurologic manifestations

of the coronaviruses in the age of coronavirus

disease 2019: a review. JAMA Neurol 2020; 77:1018-

27. [CrossRef]

6. Iadecola C, Anrather J, Kamel H. Effects of COVID-19

on the nervous system. Cell 2020; 183:16-27.e1.

[CrossRef]

7. Mao L, Jin H, Wang M, Hu Y, Chen S, He Q, et al. Neurologic

manifestations of hospitalized patients with coronavirus

disease 2019 in Wuhan, China. JAMA Neurol

2020; 77:683-90. [CrossRef]

8. Efe IE, Aydin OU, Alabulut A, Celik O, Aydin K. COVID-

19− associated encephalitis mimicking glial tumor.

World Neurosurg 2020; 140:46-8. [CrossRef]

9. Farhadian S, Glick LR, Vogels CB, Thomas J, Chiarella J,

Casanovas-Massana A, et al. Acute encephalopathy with

elevated CSF inflammatory markers as the initial presentation

of COVID-19. BMC Neurol 2020; 20:248.

[CrossRef]

10. Huang C, Wang Y, Li X, Ren L, Zhao J, Hu Y, et al. Clinical

features of patients infected with 2019 novel coronavirus

in Wuhan, China. Lancet 2020; 395:497-506.

[CrossRef]

11. Moriguchi T, Harii N, Goto J, Harada D, Sugawara H,

Takamino J, et al. A first case of meningitis/encephalitis

associated with SARS-Coronavirus-2. Int J Infect Dis

2020; 94:55-8. [CrossRef]

12. Meppiel E, Peiffer-Smadja N, Maury A, Bekri I, Delorme

C, Desestret V, et al. Neurologic manifestations associated

with COVID-19: a multicentre registry. Clin Microbiol

Infect 2021; 27:458-66. [CrossRef]

13. de Oliveira FAA, Palmeira DCC, Rocha-Filho PAS.

Headache and pleocytosis in CSF associated with

COVID-19: case report. Neurol Sci 2020; 41:3021-2.

[CrossRef]

14. Bernard‐Valnet R, Pizzarotti B, Anichini A, Demars Y,

Russo E, Schmidhauser M, et al. Two patients with acute

meningoencephalitis concomitant with SARS ‐CoV ‐ 2

infection. Eur J Neurol 2020; 27:e43-e44. [CrossRef]

15. Filatov A, Sharma P, Hindi F, Espinosa PS. Neurological

complications of coronavirus disease (COVID-19): encephalopathy.

Cureus 2020; 12:e7352. [CrossRef]



OLGU SUNUMU

CASE REPORT

Atypical Post-COVID Sequel: Bronchiectasis

Atipik Post-COVID Sekeli: Bronşektazi

Emine Afşin


Abstract

Although there have been many studies determining

the occurrence of post-COVID pulmonary fibrosis

and thromboembolism, there are a limited number of

studies and case reports in literature on the development

of bronchiectasis. The present study presents a

case of bronchiectasis sequel in the post-COVID 11th

month. A 49-year-old male, non-smoker with diabetes

mellitus and hypertension was admitted with exertional

dyspnea. The patient had been followed up in

the hospital 11 months earlier for 1.5 months with

severe COVID-19 pneumonia and respiratory failure

for which he was treated with Favipiravir, pulse

methylprednisolone and broad-spectrum antibiotics.

There was no need for invasive mechanical ventilation,

and no secondary bacterial infection was detected.

Compared to the previous CT, a chest CT

revealed that bronchiectasis had persisted despite the

disappearance of fibrotic changes. In the coming

years, one of the first questions raised regarding the

etiology of bronchiectasis may be the patient’s

COVID-19 history.

Key words: COVID- 19, bronchiectasis, post-COVID

sequel.

Öz

Post- COVID pulmoner fibrozis ve tromboemboli

gelişimine dair çok sayıda yayın olmasına rağmen,

bronşektazi gelişimi ile ilgili literatürde sınırlı sayıda

yayın ve olgu sunumları bulunmaktadır. Biz de bu

çalışmamızda post-COVID 11. ayda bronşektazi

sekeli saptanan olgumuzu sunmayı amaçladık. Kırk

dokuz yaşında, erkek, diabetes mellitus ve hipertansiyon

tanılı, non-smoker hasta, efor dispnesi ile başvurdu.

On bir ay önce ağır COVID-19 pnömonisi ve

solunum yetmezliği ile hastanede 1,5 ay süreyle yatırılarak

izlenmişti. Favipiravir, pulse metilprednizolon

ve geniş spekturumlu antibiyotik verilen hastanın

invazif mekanik ventilatör ihtiyacı olmamıştı ve sekonder

bakteriyel enfeksiyon saptanmamıştı. Toraks

BT‘sinde eski BT’leri ile kıyaslandığında fibrotik değişiklikler

kaybolmasına rağmen bronşektazisinin sebat

ettiği görüldü. Gelecek yıllarda bronşektazi etyolojisinde

ilk sorgulayacağımız nedenlerden biri COVID-

19 geçirme öyküsü olabilir.

Anahtar Sözcükler: COVID- 19, bronşiektazi, post-

COVID sekeli.

Bolu Abant İzzet Baysal University İzzet Baysal Training and Research

Hospital, Bolu, Türkiye

Bolu Abant İzzet Baysal Üniversitesi İzzet Baysal Eğitim ve

Araştırma Hastanesi, Bolu


Correspondence (İletişim): Emine Afşin, Bolu Abant İzzet Baysal University İzzet Baysal Training and Research Hospital, Bolu, Türkiye

e-mail: emineafsin@yahoo.com

112


Bronchiectasis is indicated by the inner diameter of the

bronchus being larger than the accompanying vessel on

computed tomography (CT) (bronchus/arterial ratio

greater than 0.7), by the disappearance of bronchial

narrowing or by the appearance of bronchi within 1 cm

of the pleural surface (1), or by the permanent and abnormal

dilatation of the bronchi resulting from the destruction

of elastic tissues and muscles (2,3).

While immunodeficiency syndromes, and genetic and

metabolic defects rank in the first place in the etiology in

developed countries, bacterial, viral and fungal infections

are at the forefront in developing countries (4). Respiratory

infections in childhood are usually severe. Viral infections

lead to mucociliary clearance damage, thereby

allowing the infection of the respiratory tract. Continued

infection leads to the prolongation of the inflammatory

process and bacterial colonization, leading to a repetitive

cycle that triggers progressive lung damage. With the

release of elastase, metalloproteinases and reactive oxygen

species by neutrophils, it damages elastin and basement

membrane collagen, and proteoglycans are involved

in the weakening of the bronchial wall and bronchial

enlargement (5). Elastase causes epithelial cell

damage, goblet cell hyperplasia and mucosal hypersecretion

(6).

Although viral agents are mentioned in many studies, the

effect of Coronavirus Disease 2019 (COVID-19) on the

development of bronchiectasis remains unclear. Post-

COVID chronic cough, fibrotic lung disease, pulmonary

vascular diseases and bronchiectasis have all been defined

as potential respiratory problems (7). Although

many publications have determined the occurrence of

post-COVID pulmonary fibrosis and thromboembolism,

there have been a limited number of studies and case

reports to date assessing the development of bronchiectasis

(8). The present study presents a case of bronchiectasis

sequel in the post-COVID 11th month.

respiratory failure 11 months earlier. A chest X-ray revealed

diffuse ground-glass infiltration (Figure 1a). Diffuse

ground-glass consolidations, including an air bronchogram,

were observed on chest CT taken in the post-

COVID first month (Figure 1b, c and d). The patient's file

revealed that the patient had been treated with Favipiravir,

low molecular weight heparin, piperacillin-tazobactam

and 1gr/day methylprednisolone (withdrawn and reduced)

therapy for three days along with high-flow nasal oxygen

therapy, and an oxygen concentrator was prescribed for

home use at the time. No secondary bacterial infection

agents grew in the sputum culture. The patient had no

history of being treated for pneumonia or tuberculosis

other than COVID-19, including in childhood; and there

was no chronic cough or sputum complaint. A chest CT

obtained at the post-COVID 11th month revealed bilateral

bronchiectasis, peripheral air cyst in the left lung and

peripheral atelectasis bands. Significant regression in

fibrotic appearance (Figure 2) was noted when compared

to the chest CT performed in the post-COVID third month.

The patient was informed about the necessity of flu,

pneumococcal and COVID-19 vaccines and was followed

up.

DISCUSSION

Bronchiectasis can occur rapidly and cause sequel in

cases of COVID-19 infection, and comorbidities and

secondary infections may be predisposing factors for

bronchiectasis (5). The presented case had diabetes

mellitus and hypertension, and so blood sugar regulation

may have been impaired during the period of steroid

therapy. Despite the predisposition to secondary infections

after high-dose steroids, no growth was detected in

the patient’s sputum culture.

CASE

A 49-year-old male patient was admitted with a complaint

of exertional dyspnea. A physical examination revealed

no pathological findings except for crepitation

sounds in the auscultation of the thoracic baselines. Partial

oxygen saturation in room air was 94%. The patient,

who had been diagnosed previously with diabetes mellitus

and hypertension, had no smoking history, and had

been hospitalized for approximately 1.5 months due to

severe COVID-19 pneumonia (SARS-CoV2 PCR test obtained

with nasopharyngeal sampling was positive) and

Figure 1: Chest X-ray at the time of hospitalization revealing bilateral

ground-glass infiltration (a), in the 1st month post-COVID, revealing a

peripheral air cyst in the left upper lobe, diffuse ground-glass and consolidated

areas, and bronchiectasis in the axial sections of a chest CT

(b,c,d)


Atypical Post-COVID Sequel: Bronchiectasis | Afşin et al.

COVID-19 cases with severe pneumonia and respiratory

failure.

CONCLUSION

Apart from post-COVID fibrosis and pulmonary thromboembolism,

the present study draws attention also to isolated

bronchiectasis. One of the first questions posed

regarding the etiology of bronchiectasis in the years to

come may be the patient’s COVID-19 history.


None declared.

Figure 2: Chest CT axial sections showing persistent bronchiectatic

changes despite persistent regression in fibrotic findings at the third

month post-COVID (a,c,e) and 11th month post-COVID (b,d,f)

High-resolution CT findings 3 months after discharge in

China have revealed interstitial thickening (27.27%), pure

ground glass opacity (7.27%) and crazy paving (5.45%)

findings (9), and traction bronchiectasis secondary to

post-COVID fibrosis is also common. Traction bronchiectasis

is a subtype of bronchiectasis in which the bronchi

become dilated secondary to mechanical traction due to

fibrosis of the adjacent lung parenchyma, and lung injury

resulting from invasive mechanical ventilation may also

contribute to the process. It is not known how much of the

bronchiectasis persists following the resolution of interstitial

pneumonia. Enlarged or convoluted bronchi lose their

ability to clear mucus effectively and may predispose the

patient to recurrent infections (10). Although fibrotic

changes were observed in our patient's post-COVID 3rdmonth

chest CT, most had regressed by the 11th month,

although the bronchiectasis image persisted.

Bronchiectasis associated with COVID-19 is an atypical

finding. In a retrospective study, bronchiectasis changes

were described in one of 121 COVID-19 patients (11).

Secondary bacterial infections, prolongation of weaning

from mechanical ventilation, and length of hospital stay

may lead to the formation of bronchiectasis (12). Our

patient did not need invasive mechanical ventilation during

his prolonged hospital stay of 1.5 months.

Increased interleukin-6 has been associated with the

incidence of severe bronchiectasis in tuberculosis patients

(13). Interleukin-6 is an acute phase reactant that forms

in the early phase of inflammation, and high Interleukin-6

levels have also been associated with poor prognosis in

COVID-19 (14). Based on this relationship, we suggest

that bronchiectasis may be seen more frequently in


Concept - E.A.; Planning and Design - E.A.; Supervision -

E.A.; Funding - E.A.; Materials - E.A.; Data Collection

and/or Processing - E.A.; Analysis and/or Interpretation -

E.A.; Literature Review - E.A.; Writing - E.A.; Critical Review

- E.A.

YAZAR KATKILARI

Fikir - E.A.; Tasarım ve Dizayn - E.A.; Denetleme - E.A.;

Kaynaklar - E.A.; Malzemeler - E.A.; Veri Toplama

ve/veya İşleme - E.A.; Analiz ve/veya Yorum - E.A.; Literatür

Taraması - E.A.; Yazıyı Yazan - E.A.; Eleştirel İnceleme

- E.A.

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Floto AR, et al. British Thoracic Society guideline for

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[CrossRef]

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olgusu. Van Tıp Dergisi 2011; 18: 45-8.

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RF. Bronchiectasis as A Sequelae From COVID-19.

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(ICoSIHSN 2020). 2021;33:105-8. [CrossRef]



6. King P. The pathophysiology of bronchiectasis. Int J

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BMJ 2020; 370:m3001. [CrossRef]

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