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Acquired trachea-esophageal/broncho-esophageal (TEF/BEF) fistula is a rare entity in adults, having malignant and benign causes. The commonest cause of malignant TEF is carcinoma esophagus.¹ Causes of benign TEF include intubation, trauma, surgery, and granulomatous infections. TEF is a rare consequence of tuberculosis, seen in less than 20% of patients with pulmonary tuberculosis.²

The pathogenesis of aero-digestive fistula is usually an extension of a tubercular lesion from lungs, mediastinal lymph nodes, or vertebra into the esophagus, rather than primary esophageal tuberculosis.³

The management of TEF/BEF depends on the cause, haemodynamic stability and nutritional status, and the facilities available at the treating hospital. Although surgery has been considered a primary treatment modality, TEF/BEF in tuberculosis has a variety of options available including medical management, anduse of endoscopic procedures such as stents or clips. 

This case report describes the clinical course of a patient with BEF following pulmonary tuberculosis who was initially managed with a trial of ATT (anti-tubercular therapy), and esophageal stenting, who eventually required esophagectomy. 

This case report aims to describe aero-digestive fistula as a rare complication following pulmonary tuberculosis and the options available for treatment. It highlights the importance of tailoring therapy in accordance to the nutritional and hemodynamic status of the patient, facilities available and response to treatment. 

21 year female presented with repeated episodes of low grade fever over 3 months with associated history of cough on swallowing liquids. She had additional complaints ofintermittent diarrhea. She had been treated for typhoid fever with antibiotics and antipyretics.The patient gradually developed an exacerbation of symptoms of coughfollowing intake of liquids. There was history of recent exposure to an open case of tuberculosis.

A positive sputum for AFB confirmed the diagnosis of pulmonary tuberculosis. CECT findings were suggestive ofmiliary tuberculosis with necrotic mediastinal and paratracheal lymph nodes,with a fistulous communication between the right main bronchus and the esophagus (Figure1). The patient was subsequently started on ATT. After one month of ATT, shehad persistent symptoms of cough following food intake. An upper GI endoscopy confirmed the presence of esophageal fistulous opening at 25 cm from incisors and anElla-Dannis partially covered stent was placed.

The patient initially improved symptomatically, however 3 months following stent placement she began experiencing retrosternal pain with dysphagia to solids due to stent impaction.

An upper GI endoscopy showed tissue in-growth at both ends of the stent. Endoscopic removal of the stent failed on two occasions. Hence the patient was planned for surgery.

A preoperative assessment for potential risk factors showed poor oral intake and a BMI of 17 kg/m2. High protein feeds raisedher albumin to 4.  

Repeat CT chest showed esophageal stent in situ, multiple enlarged paratracheal para aortic lymph node and paraesophageal fat stranding. There was no evidence of communication between esophagus and bronchus.Bronchoscopy done showed no fistulous opening in the right main bronchus. The patient underwent surgery to extract the stent and repair the BEF. 

Patient underwent right postero-lateral thoracotomy for impacted stent removal,esophagectomy and gastric conduit. Intraoperatively the esophagus was inflamed, dilated, friable with dense adhesions to the aorta and trachea, with an impacted stent. The stent was extracted via anesophagotomy. Healed fistula site was identified in the right main bronchus (Figure 2). The entire esophagus was resected leaving a ring of esophageal tissue abutting the fistula site. 

The patient had a stormy postoperative course with anastomotic leak on the 9th postoperative day which was managed conservatively. Patient resumed a normal diet by the 19th postoperative day.

At one year following surgerythe patient has a normal oral diet and leads a normal lifestyle having completed her course of ATT. 

India faces a heavy disease burden of   2.2 million new cases of tuberculosis, annually.⁴ Pulmonary tuberculosis results inmultiple complications including pleural effusion, empyema, bronchiectasis. TEF/BEF is a rare occurrence but isassociated with high morbidity.⁵

This case report helps demonstrate the various treatment options available for aero-digestive fistula due to tuberculosis. Case reports have described complete healing of TEF with ATT without endoscopic or surgical interventions.⁶ We are reporting in this case a patientin whom a trial of ATT for 2 months failed to improve symptoms of the patient resulting in the need for more invasive procedures.

Endoscopic management may be done using over the scope clips or stents. In this case a self-expanding partially covered metal stent was placed at the level of the fistulous opening and achieved a closure of the fistulous opening. Chauhan et al have described the use of covered stent for rapid closure of TEF with a reported closure in 89% of patients (range of 67% to 100%.). Uncovered metal stents were found to result in tissue ingrowth causing dysphagia.^7,8

Stent placement is associated with complications including displacement and impaction. The optimal time to remove an esophageal stent has no consensus, though esophageal healing usually occurs within 4 weeks of stent placement.⁸ Prolonged stent placement causes inflammation and complications including epidural abscess, aorto-esophageal fistula and stent impaction. In this patient stent was left in situ for 12 weeks which resulted in tissue ingrowth leading to dysphagia. 

This case report also demonstrates the surgical approaches that can be considered in a patient. The repair of the TEF can be done by primary closure, use of body tissues 9 or use of prosthetic materials such as Dacron or PTFE to buttress the fistulous opening. In this patient a rim of the native esophagus was left behind at the fistulous opening in the right bronchus. 

In conclusion the management of acquired TEF/BEF has multiple modalities that has tobe tailor made in accordance with the patients’ nutritional status, size of the defect, facilities available. Use of endoscopic methods should be done keeping in mind the possible complications that may follow.