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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 6  |  Issue : 1  |  Page : 53-55

Tubercular esophagocutaneous fistula: A rare case


Department of Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh, India

Date of Web Publication15-Feb-2017

Correspondence Address:
Afzal Anees
Department of Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh - 202 002, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-344X.200203

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  Abstract 

Tubercular esophagocutaneous fistula is a very rare complication arising either due to erosion by caseating cervical lymph nodes or due to tuberculosis (TB) of the esophagus. Both of these conditions are themselves rather sporadic. A young male presented with a passage of “orally taken liquids” from an opening in the neck. He had no primary foci of TB detected whatsoever, which was unusual. He was started on antitubercular treatment (ATT) for 8 weeks, but due to nonclosure of tract, the fistula had to be excised. Histopathology confirmed the tubercular nature of tract. ATT was continued postoperatively. There has been no recurrence even after 1½ years of follow-up.

Keywords: Antitubercular treatment, esophageal tuberculosis, esophagocutaneous fistula, fistulectomy, tuberculosis


How to cite this article:
Anees A, Singh KD, Khan MA, Khan S. Tubercular esophagocutaneous fistula: A rare case. Int J Health Allied Sci 2017;6:53-5

How to cite this URL:
Anees A, Singh KD, Khan MA, Khan S. Tubercular esophagocutaneous fistula: A rare case. Int J Health Allied Sci [serial online] 2017 [cited 2020 Sep 23];6:53-5. Available from: http://www.ijhas.in/text.asp?2017/6/1/53/200203

Tuberculosis (TB) remains among the infectious diseases with the most wide array of presentations. An exceedingly rare extrapulmonary manifestation is TB of the esophagus, occurring in <0.2% of all TB patients.[1],[2],[3],[4] It is commonly due to spread from adjacent foci (secondary)[1],[2],[3],[4] or infrequently can occur without any apparent secondary focus (primary).[5],[6],[7] Among the extremely uncommon complications of esophageal TB,[1],[2],[3],[4] or of a caseating eroding cervical lymph node TB,[8] is esophagocutaneous fistula (ECF). Very few such cases have been described in the literature.[8],[9],[10],[11] Our case was unique in the sense that there were no apparent primary tubercular foci at presentation.


  Case Report Top


A 16-year-old male of average built presented with a passage of only “orally taken liquids,” from a small opening in the neck for 2 months. The complaint started with a small swelling in the neck region. The patient developed discharge from the swelling after few days, and then, he recognized extravasation of small amount of “orally taken liquids” from an opening at the same location. There was no history of chronic cough, decreased appetite, evening rise of temperature, trauma to neck, previous radiation exposure, neoplasm, dysphagia, or odynophagia. On examination, there was a minute opening at the apex of left carotid triangle of the neck, with slight discharge on pressing the surrounding area. On giving milk to the patient orally, small droplets of milk immediately appeared at the mouth of the opening. Cervical lymph nodes were not enlarged, and the examination of rest of the neck and chest was within normal limits. On investigation, erythrocyte sedimentation rate (ESR) was raised and there was lymphocytosis. Mantoux test of the patient was positive. Culture of the discharge from opening was sterile and stained negative for acid-fast bacilli (AFB). X-ray of the chest and contrast-enhanced computed tomography (CECT) of the thorax revealed no abnormal finding. The internal opening could not be detected on esophagoscopy. When the patient was given methylene blue dye orally, it immediately appeared at the mouth of opening, but the tract could not be seen on fluoroscopy while using water-soluble contrast. The patient was seronegative for human immunodeficiency virus. Scrapings of tissue from the opening were subjected to fine needle aspiration cytology which showed tubercular granulomas. He was started on antitubercular treatment (ATT). Treatment continued for 2 months, but passage of orally taken liquids from the opening continued. A decision to excise the tract was taken. At the time of operation, to delineate the tract, patient was made to drink diluted methylene blue dye [Figure 1]. A fistulectomy was performed under general anesthesia while preserving vital structures in the neck [Figure 2] and making sure that the dye did not spill out in the operative field. Tract was excised and the esophageal opening was closed primarily with polyglactin 3-0 RB suture. Skin was closed with skin stapler after inserting a mini-vacuum drain. A nasogastric tube was put and feeding was started from it from the 2nd day. On the 10th day, a gastrografin study was performed which showed no spillage of contrast. Nasogastric tube was taken out and the patient was orally allowed. He was discharged on ATT. Histopathological examination (HPE) report suggested tubercular nature of excised tract [Figure 3]. Patient's ATT was continued for 9 months and he has no recurrence or complication even after 1½ years.
Figure 1: Demonstration of tract by giving patient methylene blue dye orally which is coming out from the fistulous tract in the neck

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Figure 2: Peroperative image of fistulous tract, communicating with esophagus, being excised

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Figure 3: Histopathological slide ×400 granulomas with giant cells marked with arrows and the surrounding lymphocytic infiltration

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  Discussion Top


Esophageal TB is a rare entity with an estimated prevalence of <0.2%.[1],[2],[3],[4] It is most commonly secondary to TB of lungs, mediastinum, larynx, pharynx, spine, and lymph nodes. It commonly involves the proximal rather than the distal esophagus.[1],[2],[3],[4] However, primary esophageal TB has also been reported in the literature.[5],[6],[7] The presenting clinical features of esophageal TB, in addition to the constitutional symptoms of TB, are dysphagia, epigastric pain, odynophagia, and occasionally, hematemesis.[1],[2],[3] Tubercular ECF is a very rare complication that may occur secondary to esophageal TB or an eroding caseating tubercular cervical lymph node.[8] There were no such complaints in our patient. Furthermore, there was neither history nor evidence of tubercular involvement of the lung parenchyma, pleura, or mediastinal/cervical lymph nodes in our case. Our case seems to be secondary to primary esophageal TB rather than a sequela of erosion by a caseating tubercular cervical lymph node.

Chest radiograph, esophagoscopy, contrast esophagogram, fistulogram, and CECT of the chest are important modalities to establish the diagnosis. In our case, culture was negative and no AFB organism could be demonstrated in the discharge from fistula. The internal opening of the tract could not be identified on esophagoscopy. The tract could neither be demonstrated on luminal contrast study nor a fistulogram be performed due to very small size of external opening. However, the tract was delineated using diluted methylene blue dye during surgical exploration. In our case, the basis of high suspicion for tract being tubercular in nature was the chronicity of the fistulous tract, high prevalence of TB in the region, no obvious cause, lymphocytosis with raised ESR, and positive Mantoux test.

A trial ATT for 2 months was given to the patient as esophageal TB has been reported to respond well to medical treatment in 4–8 weeks,[4],[8] even the sinuses and fistulae can heal well with ATT. However, the tract did not close even after 2 months of ATT. Now, for those who fail medical treatment, surgical exploration and excision of the tract are the definitive treatment. The case reported by Yokoyama et al.[10] and Agarwal et al.[11] also required surgery as definitive therapy. Hence, a decision to operate the patient was taken and the tract was excised. Nasogastric tube was put for at least 10 days as late leak in the esophagus is a documented complication. ATT was continued following surgery, and HPE report further supported our theory.


  Conclusion Top


Our case occurred as a sporadic event without any evidence of TB in any other part of the body; however, the chronicity of disease was typical of the tubercular course of disease. Thus, a patient presenting with an ECF without any apparent cause in a developing country like India, with high prevalence of TB, should be considered tubercular until unless proved otherwise and treated with the first-line ATT drugs. Surgery should be used only when the first-line ATT fails. Surgery needs to be followed by ATT on the basis of histopathological evidence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Sathiyasekaran M, Shivbalan S. Esophageal tuberculosis. Indian J Pediatr 2004;71:457-8.  Back to cited text no. 1
    
2.
Jain SK, Jain S, Jain M, Yaduvanshi A. Esophageal tuberculosis: Is it so rare? Report of 12 cases and review of the literature. Am J Gastroenterol 2002;97:287-91.  Back to cited text no. 2
    
3.
Jain SS, Somani PO, Mahey RC, Shah DK, Contractor QQ, Rathi PM. Esophageal tuberculosis presenting with hematemesis. World J Gastrointest Endosc 2013;5:581-3.  Back to cited text no. 3
    
4.
Hajong R, Topno N, Baruah AJ, Das R. Tubercular esophagocutaneous fistula. Indian J Surg 2013;75 Suppl 1:6-8.  Back to cited text no. 4
    
5.
Dhavan S, Vaiphei K, Chawla Y, Suri S, Dilawari JB. Primary tuberculosis of the esophagus. J Assoc Physicians India 1998;46:398.  Back to cited text no. 5
    
6.
Basharat M, Khokhar N. Primary esophageal tuberculosis. J Coll Physicians Surg Pak 2004;14:364-5.  Back to cited text no. 6
    
7.
Fujiwara T, Yoshida Y, Yamada S, Kawamata H, Fujimori T, Imawari M. A case of primary esophageal tuberculosis diagnosed by identification of mycobacteria in paraffin-embedded esophageal biopsy specimens by polymerase chain reaction. J Gastroenterol 2003;38:74-8.  Back to cited text no. 7
    
8.
Tiwari P, Gangopadhyay AN, Kumar V, Pandey V. Tubercular esophagocutaneous fistula in a child: Case report with review of literature. IOSR J Dent Med Sci 2014;13:112-4.  Back to cited text no. 8
    
9.
Xavier S, Kochhar R, Nagi B, Singh K. Tuberculous esophagocutaneous fistula. J Clin Gastroenterol 1996;23:118-20.  Back to cited text no. 9
    
10.
Yokoyama I, Ichihashi H, Kawahara K, Takada M, Kondo T. Esophago-pleuro-cutaneous fistula. A case report. Jpn J Surg 1984;14:139-42.  Back to cited text no. 10
    
11.
Agarwal V, Singh SK, Siddiqi MS, Joshi LM, Tandon S. Esophagopleural fistula following spontaneous rupture of traction diverticulum. Asian Cardiovasc Thorac Ann 2003;11:344-5.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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