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Year : 2014  |  Volume : 1  |  Issue : 2  |  Page : 81-83

Tuberculous bronchoesophageal fistula: A case report


1 Department of Surgical Gastroenterology, Jawaharlal Institute of Post Graduate Medical Education and Research (JIPMER), Pondicherry, India
2 Department of Radiology, Jawaharlal Institute of Post Graduate Medical Education and Research (JIPMER), Pondicherry, India
3 Department of Medical Gastroenterology, Jawaharlal Institute of Post Graduate Medical Education and Research (JIPMER), Pondicherry, India

Date of Web Publication29-Dec-2014

Correspondence Address:
Biju Pottakkat
Department of Surgical Gastroenterology, 4th Floor, Superspeciality Block, Jawaharlal Institute of Post Graduate Medical Education and Research (JIPMER), Pondicherry - 6
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2349-4220.148010

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  Abstract 

Tracheoesophageal fistula and bronchoesophageal fistula (BEF) usually result from malignancy. BEF caused due to benign conditions is rare. Here, we report a case of BEF due to tuberculosis. A 65-year-old lady presented with 15 days history of dysphagia, cough, and fever. Esophagoscopy revealed an ulcerated lesion at 22 cm. Further evaluation with contrast-enhanced computed tomography revealed mid-esophageal wall thickening, mediastinal and supraclavicular lymphadenopathy, along with BEF. Endoscopic tissue biopsy from ulcer revealed tuberculosis. The patient was put on antituberculous drugs and showed good response to therapy.

Keywords: Antituberculous therapy, bronchoesophageal fistula, tuberculosis


How to cite this article:
Bhati G, Pottakkat B, Kalayarasan R, Barathi D, Mohan P. Tuberculous bronchoesophageal fistula: A case report. Int J Adv Med Health Res 2014;1:81-3

How to cite this URL:
Bhati G, Pottakkat B, Kalayarasan R, Barathi D, Mohan P. Tuberculous bronchoesophageal fistula: A case report. Int J Adv Med Health Res [serial online] 2014 [cited 2018 Aug 16];1:81-3. Available from: http://www.ijamhrjournal.org/text.asp?2014/1/2/81/148010


  Introduction Top


A communication between the esophagus and the tracheal bronchial system is a rare and can be either congenital or acquired. Congenital fistulas are usually diagnosed in the neonatal period as respiratory distress and cyanosis during feeding. The most common cause of acquired fistula is malignancy of the esophagus or lung. Most of these fistulae are a result of communication between the upper esophagus and the trachea. Non-malignant causes of fistulae are rare. Diagnosis of a bronchoesophageal fistula (BEF) may sometimes be difficult because of an insidious and nonspecific clinical course. [1],[2],[3] In contrast to malignant fistula, tuberculous fistula usually involves the right or left main bronchus rather than the trachea. Closure of tuberculous fistula can usually be achieved with conservative measures. However, surgical repair may be required for large, complicated fistula.


  Case report Top


A 65-year-old lady was referred to us with provisional diagnosis of esophageal diverticulum and suspected esophageal malignancy. She presented with 15 days history of dysphagia for solids, productive cough, low-grade intermittent fever, and projectile vomiting. There was neither previous history nor family history of tuberculosis. She was a known diabetic and was on oral antidiabetics for the previous 7 years, but her blood sugar levels were uncontrolled. On examination, there was pallor and right supraclavicular lymphadenopathy. Bilateral fine crepitations were present on chest auscultation. On evaluation, she had a normal blood cell count. The erythrocyte sedimentation rate was 110 mm/h. Three sputum smear examinations were negative for acid-fast bacilli. She was found seronegative for HIV, hepatitis C virus (HCV), and hepatitis B virus (HBV). Esophagoscopy found a diverticulum at 20 cm from the incisors and a punched-out lesion was seen at 22 cm extending up to 25 cm with mucosal ulceration [Figure 1] and [Figure 2]. Chest X-ray showed left hilar lymphadenopathy. Contrast-enhanced computed tomogram (CT) scan of the neck, thorax, and abdomen [Figure 3] and [Figure 4] showed enlarged necrotic lymph nodes in the right supraclavicular, pretracheal, and left hilar areas. The esophageal diverticulum was visualized at T1 level. Communication was visualized between left bronchus and esophagus just below the carina. Esophageal wall thickening was visualized below the carina. The presentation in old age with short duration dysphagia, cough, and CT scan finding raised the suspicion of malignant esophagobronchial fistula. Multiple endoscopic esophageal mucosal biopsies including that from the punched-out lesion [Figure 5], [Figure 6] and [Figure 7] were suggestive of tuberculosis. A fine needle aspiration from the right supraclavicular lymph node showed features of tuberculous lymphadenitis. A diagnosis of tuberculous mediastinal lymphadenopathy with ruptured subcarinal lymph node into the left main bronchus and esophagus was considered. Patient underwent feeding jejunostomy and four-drug antitubercular therapy was initiated. Patient showed good response to drug therapy. At follow-up, her cough subsided and she was relieved of dysphagia; no lesion or communication with bronchus on esophagoscopy.
Figure 1: Esophagoscopy showing esophageal diverticulum and ulcerated esophageal mucosa

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Figure 2: Esophagoscopy showing esophageal diverticulum and ulcerated esophageal mucosa

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Figure 3: CT thorax pictures showing left bronchoesophageal fistula and esophageal wall thickening mimicking esophageal malignancy

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Figure 4: CT thorax pictures showing left bronchoesophageal fistula and esophageal wall thickening mimicking esophageal malignancy

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Figure 5: Photomicrograph of the esophagus shows stratifi ed squamous epithelial lining and the subepithelium shows multiple caseating epithelioid granulomas (H&E, 40×). Inset-Photomicrograph showing stratifi ed squamous epithelial lining with ulceration. There is an underlying caseating epithelioid granuloma comprising Langhan's giant cell along with epithelioid cells, lymphocytes, and plasma cells (H&E, 100×)

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Figure 6: Photomicrograph shows caseous necrosis (arrow) surrounded by lymphocytes and histiocytes (H&E, 100×)

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Figure 7: Photomicrograph shows acid-fast bacilli (Ziehl– Neelsen stain, 1000×)

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  Discussion Top


Acquired causes of BEF include malignancies involving the esophagus or the adjacent structures, granulomatous infections, trauma, prolonged endotracheal intubation, endoscopic procedures, impaction of foreign body, radiotherapy, and corrosive poisoning. Most of the acquired cases are malignant in origin and secondary to esophageal or lung cancer. [4] Benign causes were found to be responsible for only 5-6% of such cases. [5] BEF secondary to tuberculosis is rare. [6],[7] Lado et al., in their literature review, reported 36 cases of BEF secondary to tuberculosis from 1893 AD to 2002 AD. [8] The proposed mechanism of development of a BEF in tuberculosis is the involvement of the mediastinal lymph nodes. Inflammation involves the surrounding tracheobronchial tree and esophagus leading to either rupture of abscess with caseous necrosis or fibrous scar causing traction diverticulum of the esophagus with subsequent fistula formation at the tip of the diverticulum. In recent years, an increasing number of cases of tuberculous BEF in association with HIV infection have been reported. [9]

The symptoms and clinical presentation of tuberculous BEF can mimic malignancy of esophagus or lung as in our patient. The most common reported symptoms of tuberculous BEF are chronic paroxysmal cough, dysphagia, fever, and pneumonia. Barium esophagogram is the most sensitive method of diagnosing BEF. Endoscopy is useful for obtaining tissue for biopsy. Computed tomogram (CT) scan of the thorax allows evaluation for neoplasm, lymphadenopathy, or other associated anomalies and it may demonstrate the fistula. [3]

Traditionally the treatment of tuberculous TEF or BEF is surgical resection or ligation/suturing of the fistulous tract. Patients with recurrent pulmonary infections and bronchiectasis may require resection of the affected lung segment. [10] Medical management with antitubercular therapy and nasogastric feeds or jejunostomy feeds for patients with tubercular BEF has been reported as a successful modality of treatment. [9] The importance of high index of suspicion and early diagnosis for conservative management of tuberculous BEF cannot be overemphasized.

We conclude that having excluded the possibility of a cancer or other associated factors, the presence of BEF with intrathoracic adenopathy is highly suggestive of tuberculosis, particularly in high-incidence areas. Antituberculous chemotherapy alone may be sufficient for the management of such patients.

 
  References Top

1.
Wigley FM, Murray HW, Mann RB, Saba GP, Kashima H, Mann JJ. Unusual manifestation of tuberculosis: TE fistula. Am J Med 1976;60:310-4.  Back to cited text no. 1
    
2.
Spalding AR, Burney DP, Richie RE. Acquired benign bronchoesophageal fistulas in the adult. Ann Thorac Surg 1979;28:378-83.  Back to cited text no. 2
    
3.
Im JG, Kim JH, Han MC, Kim CW. Computed tomography of esophagomediastinal fistula in tuberculous mediastinal lymphadenitis. J Comput Assist Tomogr 1990;14:89-92.  Back to cited text no. 3
    
4.
Aggarwal D, Mohapatra PR, Malhotra B. Acquired bronchoesophageal fistula. Lung India 2009;26:24-5.  Back to cited text no. 4
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5.
Ahn JY, Jung HY, Choi JY, Kim MY, Lee JH, Choi KS, et al. Benign bronchoesophageal fistula in adults: Endoscopic closure as primary treatment. Gut Liver 2010;4:508-13.  Back to cited text no. 5
    
6.
Sasaki M, Mochizuki H, Takahashi H. A bronchoesophageal fistula that developed shortly after the initiation of antituberculous chemotherapy. Intern Med 2013;52:795-9.  Back to cited text no. 6
    
7.
Liao LY, Wu H, Zhang NF, Liu CL, Li SY, Gu YY, et al. Bronchoesophageal fistula secondary to mediastinal lymph node tuberculosis: A case report and review of the literature. Zhonghua Jie He He Hu Xi Za Zhi 2013;36:829-932.  Back to cited text no. 7
    
8.
Lado Lado FL, Golpe Gómez A, Cabarcos Ortíz de Barrón A, Antúnez López JR. Bronchoesophageal fistulae secondary to tuberculosis. Respiration 2002;69:362-5.  Back to cited text no. 8
    
9.
Porter JC, Friedland JS, Freedman AR. Tuberculous bronchoesophageal fistulae in patients infected with the human immunodeficiency virus: Three case reports and review. Clin Infect Dis 1994;19:954-7.  Back to cited text no. 9
    
10.
Kim HK, Choi YS, Kim K, Kim J, Shim YM. Long-term results of surgical treatment in benign bronchoesophageal fistula. J Thorac Cardiovasc Surg 2007;134:411-4.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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