Tracheoesophageal Fistula as a Rare Cause of Persistent Pulmonary Tuberculosis Symptoms

Abstract

Tracheoesophageal fistula (TEF) is a rare yet severe complication of tuberculosis (TB), often leading to persistent symptoms, recurrent infections, and treatment failure. Its early recognition and appropriate management are crucial to improving patient outcomes. We report the case of a 24-year-old woman with pulmonary TB who presented with persistent fever, copious expectoration, and progressive weight loss despite receiving anti-tubercular therapy (ATT) for 3 months. She developed a right pneumothorax, requiring Malecot catheter insertion, and was referred to our institute due to poor treatment response. Differential diagnoses, including drug-resistant TB and malabsorption, were ruled out. Clinical suspicion of TEF arose due to worsening cough on swallowing. Bronchoscopy confirmed a TEF, five tracheal rings above the carina, measuring 7–8 mm in diameter. A computed tomography scan delineated its extent, and bronchoalveolar lavage analysis confirmed Mycobacterium TB. The patient was managed with nasogastric feeding, intravenous antibiotics, and ATT continuation. Her condition improved significantly within 10 days. TEF is a rare but serious complication of pulmonary TB that can significantly impact treatment success. Early recognition, nutritional support, and ATT continuation are crucial for improved outcomes and preventing complications this case underscores the importance of considering TEF in patients with persistent symptoms despite standard TB treatment.

INTRODUCTION

Pulmonary tuberculosis (TB) continues to pose a significant health challenge in India, with over 2.5 million cases reported each year (India TB Report 2023). Despite advancements in TB management, treatment failure remains a concern, occurring in up to 15% of cases. Treatment failure can result from a variety of factors, including poor adherence to therapy, underlying malnutrition, socioeconomic disparities, immune-compromised states, and drug-resistant TB (DR-TB).[1]

While systemic and patient-related factors often dominate discussions surrounding treatment failure, structural complications – though relatively rare – may play a critical and often overlooked role in impairing treatment outcomes. These complications, when unrecognized, can perpetuate symptoms, delay recovery, and increase morbidity. Such factors can include thick-walled cavity, extensive pulmonary disease and disseminated disease.[2,3]

Trachoesophageal fistula (TEF), characterized by abnormal communication between the trachea and esophagus, is an unusual manifestation of pulmonary TB seen in <10% of cases. It often presents with symptoms such as persistent cough, recurrent aspiration, dysphagia, and poor general condition, which can mimic other advanced forms of the disease or alternative pathologies. TEF is particularly challenging as it can lead to persistent or recurrent infections, worsening nutritional status, and failure of standard antitubercular therapy (ATT).[4,5]

In this case report, we discuss a rare presentation of pulmonary TB complicated by a TEF, which contributed to a poor treatment response. The case highlights how timely recognition and targeted management of structural complications, such as TEF, can dramatically alter the disease trajectory.

CASE REPORT

A 24-year-old woman from Alwar, Rajasthan, presented with persistent fever, profuse expectoration, and progressive weight loss despite receiving ATT initiated on clinical grounds 3 months earlier. The patient had developed a right pneumothorax, for which a Malecot catheter was inserted [Figure 1] before referral to our institute.

Figure 1.

X-ray and CT chest on presentation showing extensive left consolidation with right pneumothorax

Due to inadequate response to anti-TB therapy despite appropriate dosing, she was referred to our institute for further evaluation. The differential diagnoses considered included a nonresolving TB lesion, DR-TB, poor drug compliance, and poor drug absorption. However, all of these were ruled out. Her worsening cough, especially with liquid intake, heightened suspicion of a TEF. On physical examination, the patient was severely malnourished, weighing 27 kg, febrile, and exhibiting signs of respiratory distress. Laboratory investigations revealed anemia (hemoglobin [Hb]: 7.2 g/dL), leukocytosis (total leukocyte count [TLC]: 35,000/μL; neutrophils – 90%, lymphocytes – 8%, and monocytes – 2%), and significantly elevated inflammatory markers. Given her history of excessive coughing and shortness of breath while swallowing, an emergency bronchoscopy was performed to confirm the suspected fistula. This confirmed the presence of a TEF located five tracheal rings above the carina, measuring 7–8 mm in diameter [Figure 2].

Figure 2.

Bronchoscopic view of large TEF

A computed tomography (CT) scan further delineated the extent of the fistula and associated complications [Figure 3]. A mucosal biopsy obtained from the walls of the TEF revealed acute-on-chronic inflammation. Bronchoalveolar lavage (BAL) was positive for acid-fast bacilli, and the Line Probe Assay showed sensitivity to Isoniazid (H) and Rifampicin (R), microbiologically confirming the diagnosis of TB. DR-TB was ruled out based on BAL findings. BAL pyogenic culture showed pseudomonas aeruginosa, which was pan-sensitive.

Figure 3.

CT visualization of TEF

The management strategy centered on supportive care and addressing the underlying TB. A nasogastric (NG) tube was inserted to bypass the fistula, allowing adequate calorie intake. Over the hospital course, the patient gained 3 kg, improving from 27 kg to 30 kg within 10 days. Empiric intravenous antibiotics were administered, including meropenem, metronidazole, and amikacin, to target secondary infections and provide anaerobic coverage. ATT was continued through the NG tube, ensuring uninterrupted treatment despite the fistula. The right pneumothorax improved gradually with ATT [Figure 4] and resolved for which pleurodesis was done with povidone-iodine [Figure 5].

Figure 4.

X-Ray showing marked clinical improvement and resolution of right pneumothorax

Figure 5.

X-Ray post pleurodesis and drain removal, Pre-discharge

The patient demonstrated significant improvement within days. Her fever resolved by day 5, TLC decreased from 35,000/μL to 18,000/μL by day 2 and normalized to 11,000/μL by day 7, while Hb levels increased by 2 g/dL within the 1^st week. Her symptoms, including cough and aspiration, markedly subsided. After 10 days of hospitalization, she was discharged in stable condition with recommendations to continue ATT, NG feeding, and a high-calorie diet. A follow-up bronchoscopy and CT scan were scheduled for 3 months later to reassess the status of the TEF. Unfortunately, the patient has been lost to follow-up at the time of this report.

DISCUSSION

TEF is a rare but serious complication of several medical conditions, particularly TB. It presents a complex challenge for clinicians due to its significant impact on swallowing, recurrent respiratory infections, malnutrition, and overall patient survival. Acquired forms may be either benign or malignant. The causes of benign TEF include granulomatous diseases (TB/nocardia), inflammatory conditions (sarcoidosis/granulomatosis with polyangitis (GPA)), caustic alkali ingestion, prolonged ventilation, and traumatic injury.[6,7,8]

Etiopathogenesis and risk factors

TEF occurs when the shared wall between the trachea and esophagus erodes, creating an abnormal connection. In TB, this is often caused by necrotizing mediastinal lymphadenopathy, prolonged inflammation, or mechanical trauma (e.g., stenting or prolonged intubation). Rarely, traction diverticula may cause TEF. Contributing factors include poor nutritional status, immune suppression (e.g., HIV/AIDS), previous pulmonary infections, and delays in initiating TB treatment, which all result in more severe forms of infection with inflammation and necrosis. In TB-endemic areas, complications such as bronchial compression further exacerbate the condition.[5,9,10,11]

Mechanisms of tracheoesophageal fistula in tuberculosis as described in literature

Ruptured mediastinal lymph node causing track

• In many cases, caseating necrosis of TB-infected mediastinal lymph nodes leads to erosion of adjacent structures, including the trachea and esophagus

• Macchiarini et al. described a case where mediastinal adenopathy caused a large TEF

• Baijal et al. in the series of 5 cases, showed that 3 cases had necrotic lymph nodes, which may have been the focus of inflammation for TEF.[5,6]

Direct involvement of the tracheobronchial tree

• Most commonly described in literature

• Endobronchial TB can cause direct erosion of the tracheal or bronchial walls into the esophagus

• Porter et al. (1994)[9] reported bronchoesophageal fistulas associated with pulmonary TB, likely from localized necrosis and inflammation

• Rosario et al. (1996),[10] Poonam et al., Baijal et al. (2 cases), Mosquera et al., Narayanan et al. in 2017, Desai et al. in 2019[12] also described similar mechanisms of TEF.[4,5,7,9,12,13]

• Our case also describes the TEF formation by this mechanism, as evidenced by the histopathological examination of tracheal mucosa.

Granulomatous inflammation with tissue necrosis and ulceration

• Chronic granulomatous inflammation may lead to necrosis of the esophageal wall or tracheal structures without significant lymphadenopathy.[7,14]

Poststent or postsurgical erosion

• Iatrogenic causes, including stent migration or improper stent placement, can exacerbate tissue damage, leading to secondary TEF

• Guo et al. documented a stent-related TEF that necessitated further intervention.[11]

Management approaches

The treatment of TEF depends on the cause, size, and overall health of the patient. Broadly, it is categorized into conservative, endoscopic, and surgical approaches:

Conservative management

ATT remains the cornerstone of treatment for TB-related TEF. Smaller fistulas, as reported in cases by Porter and Kanarek, Yadav et al., Desai et al. Narayanan et al. resolved with ATT and bypass feeding either through NG tube or percutaneous endoscopic gastrostomy tube. For smaller defects or patients with milder symptoms, a conservative approach with close monitoring may be effective. However, this option is only feasible for stable patients with milder symptoms.[8,9,12,13]

Surgical repair

Surgery remains the preferred approach for larger or complex fistulas. Techniques such as direct closure with muscle flap reinforcement or esophageal diversion provide definitive treatment. Macchiarini’s cervical repair and the PGIMER case involving esophagectomy highlight successful surgical outcomes.[4,6] Advances in thoracoscopic methods have further reduced complications and recovery time.[14] If left untreated, TEF leads to significant morbidity and mortality due to aspiration pneumonia and severe malnutrition. Surgical or endoscopic correction typically results in symptom resolution and a marked improvement in quality of life. Surgical repair, particularly for larger fistulas, achieves closure rates exceeding 90% in most reports.[4,6] However, as Guo et al.[11] reported, surgical intervention is not without challenges, with some cases requiring multiple procedures before achieving success. In addition, surgical correction may need to be delayed until adequate nutritional rehabilitation and control of active TB are achieved.[5,11]

Endoscopic interventions

Minimally invasive methods such as stenting and the over-the-scope clip (OTSC) system have shown promise for managing smaller fistulas. Mosquera et al. reported successful closure with OTSC in a localized case. Palliative measures such as placement of Y stent in trachea (silicone) or other stents may be used to make the patient fit for definite surgical correction. However, endoscopic techniques carry risks such as stent migration or erosion, particularly in larger or friable fistulas.[7,10,11]

Our case aligns with previous reports by Baijal et al.[5] and Desai et al.,[13] where TEF occurred due to direct tracheobronchial involvement in TB. However, unlike the case described by Macchiarini et al.,[6] which necessitated surgical intervention, our patient demonstrated significant improvement with conservative measures, reinforcing the role of early supportive care. Table 1 summarizes the various reported cases of TEF in TB with their causes and management strategies.

Table 1.

Comparison of the published cases of tracheoesophageal fistula due to tuberculosis with their causes and management

Year/Author	Cause of TEF	Size	Management	Outcome
1993/Macchiarini et al.	Mediastinal lymphadenopathy	Large (single)	Surgical repair – tracheal resection	Resolved: No recurrence
1994/Porter et al.	Direct erosion from endobronchial TB	Small (multiple)	ATT; NG feeding	Healed in 9 months
1996/Rosario et al.	Extensive necrosis due to TB in AIDS	Small (single)	Tracheal stenting followed by surgical repair	Resolved
2008/Ashwitha Ravi et al.	Granulomatous inflammation and necrosis causing rupture and TEF	Large (single)	Tracheal stenting followed by esophageal resection	Resolved
2013/Baijal et al.	5 cases series - ruptured mediastinal LNs (3/5)	Large	1 patient – Glue with hemoclip, 3 patients – PEG tubes, 1 patient - conservative care	Resolved
2017/Santosh Narayanan et al.	Endobronchial extension of TB	Large	ATT with conservative management	Resolved
2018/Mosquera et al.	Tissue necrosis and tracheal damage	Small	Endoscopic correction with over the scope clip	Resolved
2019/Gucsav et al.	Tissue necrosis and tracheal damage	Small	Y-shaped tracheal stent	Symptomatic relief
2019/Desai et al.	Endobronchial extension of TB	Large	PEG tubes, conservative care	Resolved
2022/Guo et al.	Stent related erosion (stent placed for tracheal stenosis)	Large	Repeat tracheal stent/local growth factor instillation	Healed with prolonged course
2023/Yadav et al.	Esophageal ulceration due to TB	Small	ATT, conservative	Healed
2024/Saini et al.	Endobronchial extension of TB	Large	NG feeding, ATT conservative care	Symptomatic improvement

TB=Tuberculosis, TEF=Tracheoesophageal fistula, ATT=Antitubercular therapy, AIDS=Acquired immune deficiency syndrome, NG=Nasogastric, PEG=Percutaneous endoscopic gastrostomy, LNs=Lymph nodes

CONCLUSION

Early recognition and appropriate management of TEF in pulmonary TB can significantly improve patient outcomes. This case underscores the importance of considering uncommon structural complications, such as TEF, which may present as seemingly harmless symptoms of TB disease such as cough, dysphagia, and loss of appetite. In our case, prompt switch from oral to Ryles tube feeding was sufficient to cause significant improvement in the patients’ health. It also highlights the critical role of a coordinated, multidisciplinary approach in ensuring timely diagnosis and effective intervention. By maintaining awareness of these rare complications, we can improve patient outcomes and enhance the overall quality of TB care.

Declaration of patient consent

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

Conflicts of interest

There are no conflicts of interest.

Funding Statement

Nil.