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Bilateral multiple tumor-like endobronchial tuberculosis, diagnosed with bronchoscopic examination

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tuberculosis, diagnosed with bronchoscopic examination

Yasemin SAYGIDEĞER, Burcu OKTAY, Emine SEVGİ, Özlem SEVER, Hikmet FIRAT, Sadık ARDIÇ SB Dışkapı Yıldırım Beyazıt Eğitim ve Araştırma Hastanesi, Göğüs Hastalıkları Kliniği, Ankara.

ÖZET

Bronkoskopik inceleme ile tanı alan endobronşiyal tüberküloz olgusu

Endobronşiyal tüberküloz (EBTB), trakeobronşiyal ağacın tüberküloz ile infeksiyonu olarak tanımlanır ve erişkin popülas- yonda sık rastlanmayan bir durumdur. Parankimal hastalık görülmediğinde EBTB daha az fark edilmekte ve tanıda zor- luklara neden olmaktadır. Amacımız, az görülen ve seyri sırasında bronkostenoz gelişebilmesi açısından önemli olan bu hastalıkla ilgili bir olguyu sunmaktır. Olgudaki, 20 yaşındaki kadın hasta iki aydır nonprodüktif öksürük, nefes darlığı ve son dönemde minimal hemoptizi ile başvurmuştur. Klinik ve radyolojik değerlendirme sonrasında fleksibl bronkoskopi ile ana karinadan itibaren her iki ana bronşta çok sayıda tümör görünümlü endobronşiyal lezyon izlenmiştir. Biyopsi sonrası tanı EBTB gelmiş ve antitüberküloz tedavinin ikinci ayında endobronşiyal lezyonlar kaybolmuştur. Hasta stenoz gelişme- den iyileşmiştir. Bu olgu, endobronşiyal lezyonların ayırıcı tanısında EBTB’nin de akla gelmesi gerekliliğini hatırlatmakta- dır. EBTB olgularının komplikasyonsuz iyileşebilmesi zamanında tanı ve tedavi başlanması ile olanaklıdır.

Anahtar Kelimeler: Endobronşiyal tüberküloz, bronkoskopi, endobronşiyal kitle.

SUMMARY

Bilateral multiple tumor-like endobronchial tuberculosis, diagnosed with bronchoscopic examination

Yasemin SAYGIDEĞER, Burcu OKTAY, Emine SEVGİ, Özlem SEVER, Hikmet FIRAT, Sadık ARDIÇ

Department of Chest Diseases, Diskapi Yildirim Beyazit Training and Research Hospital, Ankara, Turkey.

Endobronchial tuberculosis is defined as tuberculosis infection of tracheobronchial tree and it is not seen often in adult po- pulation. In the absence of parenchymal disease endobronchial tuberculosis is less well-recognized and can lead to difficul- ties in diagnosis. Our aim is to introduce a rare form of tuberculosis that is important because of high probability of deve- loping severe bronchostenosis during its course. We report a 20-year-old woman who presented with two-month history of severe non-productive cough, shortness of breath, and hemoptysis. After clinical and radiological evaluation, flexible bronc-

Yazışma Adresi (Address for Correspondence):

Dr. Yasemin SAYGIDEĞER, SB Dışkapı Yıldırım Beyazıt Eğitim ve Araştırma Hastanesi, Göğüs Hastalıkları Kliniği, Altındağ, ANKARA - TURKEY

e-mail: ysaygideger@gmail.com

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Although endobronchial tuberculosis is a rare condition in adult population, it continuous to be a health prob- lem because of having a high incidence of positive spu- tum samples and high probability of developing severe bronchostenosis during its course (1,2). It is often mis- diagnosed as bronchial asthma and lung cancer (3-5).

Misdiagnose of the disease leads to increase public he- alth concern. Endobronchial tuberculosis is usually thought to be associated with pulmonary tuberculosis (6). However it could be occur in the absence of pa- renchymal infiltration as well (4). Early diagnosis and early administration of antituberculosis chemotherapy before involvement of the deeper airways is important to prevent the development of bronchostenosis (7). We report a patient with endobronchial tuberculosis who had no parenchymal infiltration and healed without bronchostenosis.

CASE REPORT

A 20-year-old woman presented with a two-month history of severe non-productive cough, shortness of breath and wheezing. She had noted difficulty in swallowing and described a small amount of fresh blood came after a strong and severe chough for on- ce, one week before she referred to our clinic. She had no history of bronchial asthma, previous chronic disease and had never smoked. She did not compla- in of weight loss, fever and night sweats. She had gi- ven non-specific oral antibiotics and inhaler form of bronchodilators without any improvement. She re- ported deterioration in her symptoms during previous one month. On physical examination her vital signs were stable. There was no lymphadenopathy, enlar- gement of liver or spleen. Auscultation of the chest was normal. No rale, rhoncus and cardiac murmur was detected. Her erythrocyte sedimentation rate was 56 mm/hour. Serum hemoglobin level was 9.6 g/dL, total white blood cell count was 9.10 x 109/L.

Lymphocytes were 17.2%, platelets were 405 x 109/L. Spirometric examination was normal. She co- uld not obtain available sputum sample for the exa- mination of acid-fast bacilli.

The chest radiograph showed a prominent left hilum with no infiltration on the lung parenchyma (Figure 1).

The chest computed tomography demonstrated a polyploid figure in the left main bronchus and a figure of mass on the upper part of the left hilum (Figure 2).

After her clinical history and chest computed tomog- raphy scan were evaluated, we decided to carry out fle- xible bronchoscopic examination with the suspicious of malignancy and endobronchial tuberculosis. Flexible bronchoscopy showed bilateral multiple tumorous lesi- ons that were seen from main carina down to the both main bronchus (Figure 3,4). The mucosa was mildly hyperemic and minimal edematous. The mucosal punch biopsies were obtained from the endobronchial tumorous lesions. Histopathologic examination reve- aled granulomatous inflammation without caseificati- on, but compatible with tuberculosis. Special stains showed no acid-fast bacilli and no malignant cells. On the basis of clinical, radiologic and histopathologic fin- dings, anti-tuberculosis therapy initiated. The patient was treated with isoniazid, rifampicin, morphzinamid and ethambutol. Neither oral nor inhaler corticosteroid was given.

Saygıdeğer Y, Oktay B, Sevgi E, Sever Ö, Fırat H, Ardıç S.

Figure 1. Chest radiograph demonstrates a prominent left hi- lum but no pulmonary infiltration.

hoscopy showed bilateral multiple tumorous lesions that were seen from main carina down to the both main bronchus.

The biopsy samples revealed EBTB diagnosis and antituberculosis therapy was given. At the second month of the therapy, rebronchoscopy revealed almost disappearance of the polypoid lesions. The patient healed without any stenosis. This ca- se report is a reminder that endobronchial tuberculosis must take into consideration in differential diagnosis of endobronc- hial lesions. In patients with endobronchial tuberculosis healing without any complication could be achieved with timely diagnosis and commencement of early treatment.

Key Words: Endobronchial tuberculosis, bronchoscopy, endobronchial mass.

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The culture of bronchial lavage at four weeks was re- ported negative for tuberculosis. She reported resoluti- on of her symptoms after one month of therapy. At the second month of the therapy, rebronchoscopy revealed almost disappearance of the tumorous lesions (Figure

5,6). Therapy was prolonged for six months with isoni- azid and rifampycin. The patient healed successfully without bronchostenosis.

Figure 3. Bronchoscopic image of the main carina and the bi- lateral polypoid lesions throught both left and right main bronchus.

Figure 4. Bronchoscopic image of the left main bronchus and polypoid lesions.

Figure 5. Second month of treatment, brochoscopic image of main carina.

Figure 6. Second month of treatment, bronchoscopic image of left main bronchus.

Figure 2. Chest computed tomography demonstrates polypoid figure in the left main bronchus and a figure of mass on the upper part of the left hilum.

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DISCUSSION

Endobronchial tuberculosis is known as a very infecti- ous form of tuberculosis that remains a diagnostic challenge (8). It is usually seen at the younger popula- tion, and female predominance (3). The pathogenesis of endobronchial tuberculosis is not yet fully establis- hed. However, proposed mechanisms include direct implantation of tubercle bacilli into the broncus from an adjacent pulmonary parenchymal lesion, direct airway infiltration from an adjacent tuberculous mediastinal lymph node, erosion and protrusion of an intrathoracic tuberculous lymph node into bronchus, hematogenous spread, and extension to the peribronchial region by lymphatic drainage (2,3,9).

Clinical manifestations of endobronchial tuberculosis are non-specific and include chronic productive co- ugh, barking cough, chest pain, haemoptysis, genera- lized weakness, dyspnoea and fever (10). 10% to 20%

of patients with endobronchial tuberculosis may have normal chest radiographs. Because of these reasons diagnose is often delayed and/or patients are misdi- agnosed as bronchial asthma and malignancy (4).

Delayed diagnose leads to delayed treatment which is associated with increased complication rate and pub- lic health concern (7). The most important goals of the treatment in endobronchial tuberculosis are the eradication of tubercle bacilli and prevention of bronchial stenosis (11). Bronchial stricture could de- velop in 60-95% of cases (12,13). Computed tomog- raphy is very useful in evaluating endobronchial lesi- ons such as obstruction and stenosis (14). In our ca- se, endobronchial tumorous lesions were detected by computed tomography. At this point it is important to take into consideration endobronchial tuberculosis in the differential diagnosis of endobronchial lesions.

The bronchoscophic approach is mandatory to prompt diagnosis of endobronchial tuberculosis (15,16). Chung and Lee, described the bronchoscopic features of endobronchial tuberculosis and classified it into seven subtypes as;

1. Actively caseating, 2. Oedematous-hyperaemic, 3. Fibrostenotic,

4. Tumorous, 5. Granuler, 6. Ulcerative,

7. Non-spesific bronchitic endobronchial tuberculosis.

They also defined the therapeutic outcomes of these subtypes (2).

In our case, bilateral, multiple tumorous lesions were seen on bronchoscopic examination. The tumorous le- sions were minimally obstructing the bronchus and se- emed to not covered with caseous material as opposed to tumorous endobronchial tuberculosis. Only mild mu- cosal swelling and/or hyperemia were seen on bronc- hoscopy. Bronchoscopic and computed tomography images of the lesions were simulating malignancy. Ho- wever tuberculosis was proven by bronchoscopic bi- opsy of these lesions. At the second month of the the- rapy, rebronchoscopy revealed the almost disappearan- ce of the tumorous lesions. The prognosis was so excel- lent. Although mild mucosal swelling and/or hyperemia and good prognosis were compatible with non-spesific bronchitic endobronchial tuberculosis, we did not clas- sified our patient into this type. Because in our case ad- dition to mild mucosal swelling and/or hyperemia, the- re were multiple bilateral tumorous lesions on bronc- hoscophy. According to current classification of endob- ronchial tuberculosis, our case did not correspond clini- cally with any of seven types similar to Al-Maslamani’s case, and our case did not show any bronchostenosis si- milar to Araz’s study (17,18). Considering the recently reported cases it may suggest that future researches are required to create more comprehensive classification of endobronchial tuberculosis.

In conclusion, this case report is a reminder that en- dobronchial tuberculosis must take into consideration in differential diagnosis of endobronchial lesions. In pa- tients with endobronchial tuberculosis healing without any complication could be achieved with timely diag- nosis and commencement of early treatment.

CONFLICT of INTEREST None declared.

REFERENCES

1. Kreisel D, Arora N, Weisenberg S, Saqi A, Krupnick A, Demet- riades A, et al. Tuberculosis presenting as an endobronchial mass. J Thorac Cardiovasc Surg 2007; 133: 582-4.

2. Chung H, Lee J. Bronchoscopic assessment of the evolution of endobronchial tuberculosis. Chest 2000; 117: 385-92.

3. Lee J, Park S, Lee D, Shin D, Yang S, Yoo B. Endobronchial tuber- culosis. Clinical and bronchoscopic features in 121 cases. Chest 1992; 102: 990-4.

4. Lee T, Sin Fai, Lam K. Endobronchial tuberculosis simulating bronchial asthma. Singapore Med J 2004; 45: 390-2.

5. Matthews J, Matarese S, Carpenter J. Endobronchial tubercu- losis simulating lung cancer. Chest 1984; 86: 642-4.

6. Hoheisel G, Chan B, Chan C, Chan K, Teschler H, Costabel U.

Endobronchial tuberculosis: Diagnostic features and therape- utic outcome. Respir Med 1994; 88: 593-7.

Saygıdeğer Y, Oktay B, Sevgi E, Sever Ö, Fırat H, Ardıç S.

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7. Um S, Yoon Y, Lee S, Yim J, Yoo C, Chung H, et al. Predictors of persistent airway stenosis in patients with endobronchial tuberculosis. Int J Tuberc Lung Dis 2008; 12: 57-62.

8. Kashyap S, Mohapatra P, Saini V. Endobronchial tuberculosis.

Indian J Chest Dis Allied Sci 2003; 45: 247-56.

9. Kim Y, Kim H, Lee K, Uh S, Cung Y, Park C. Serial fiberoptic bronchoscopic observations of endobronchial tuberculosis be- fore and early after antituberculosis chemotherapy. Chest 1993; 103: 673-7.

10. Park M, Woo I, Son J, Lee S, Kim D, Mo E, et al. Endobronchial tuberculosis with expectoration of tracheal cartilages. Eur Res- pir J 2000; 15: 800-2.

11. Rikimaru T. Endobronchial tuberculosis. Expert Rev Anti In- fect Ther 2004; 2: 245-51.

12. Chan H, Sun A, Hoheisel G. Endobronchial tuberculosis-is cor- ticosteroid treatment useful? A report of 8 cases and review of the literature. Postgrad Med J 1990; 66: 822-6.

13. Van den Brande P, Van de Mierop F, Verbeken E, Demedts M.

Clinical spectrum of endobronchial tuberculosis in elderly pa- tients. Arch Intern Med 1990; 150: 2105-8.

14. Rikimaru T. Therapeutic management of endobronchial tuber- culosis. Expert Opin Pharmacother 2004; 5: 1463-70.

15. Lee J, Chung H. Bronchoscopic, radiologic and pulmonary function evaluation of endobronchial tuberculosis. Respiro- logy 2000; 5: 411-7.

16. Cakir E, Uyan ZS, Oktem S, Karakoc F, Ersu R, Karadag B, et al. Flexible bronchoscopy for diagnosis and follow up of child- hood endobronchial tuberculosis. Ped Infec Dis J 2008; 27:

783-7.

17. Al-Maslamani M, Ibrahim WH, Chacko K, Al-Khal A. Endob- ronchial tuberculosis simulating lung cancer and healing.

Libyan J Med 2008, AOP: 080330.

18. Araz O, Akgün M, Sağlam M, S, Özden K, Mirici A. The diag- nostic value of bronchoscopy in smear negative cases with pulmonary tuberculosis. Tuberk Toraks 2008; 56: 150-7.

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