Clin Endosc.  2023 Mar;56(2):239-244. 10.5946/ce.2021.215.

Bronchoesophageal fistula in a patient with Crohn’s disease receiving anti-tumor necrosis factor therapy

• Oh K
• Choi KD
• Kim HR
• Shim TS
• Ye BD
• Yang SK
• Park SH

• KMID: 2540742
• DOI: http://doi.org/10.5946/ce.2021.215

Abstract

Tuberculosis is an adverse event in patients with Crohn’s disease receiving anti-tumor necrosis factor (TNF) therapy. However, tuberculosis presenting as a bronchoesophageal fistula (BEF) is rare. We report a case of tuberculosis and BEF in a patient with Crohn’s disease who received anti-TNF therapy. A 33-year-old Korean woman developed fever and cough 2 months after initiation of anti-TNF therapy. And the symptoms persisted for 1 months, so she visited the emergency room. Chest computed tomography was performed upon visiting the emergency room, which showed BEF with aspiration pneumonia. Esophagogastroduodenoscopy with biopsy and endobronchial ultrasound with transbronchial needle aspiration confirmed that the cause of BEF was tuberculosis. Anti-tuberculosis medications were administered, and esophageal stent insertion through endoscopy was performed to manage the BEF. However, the patient’s condition did not improve; therefore, fistulectomy with primary closure was performed. After fistulectomy, the anastomosis site healing was delayed due to severe inflammation, a second esophageal stent and gastrostomy tube were inserted. Nine months after the diagnosis, the fistula disappeared without recurrence, and the esophageal stent and gastrostomy tube were removed.

Keyword

Crohn’s disease; Fistula; Tuberculosis; Tumor necrosis factor inhibitors

Figure

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  Fig. 1. (A, B) Chest computed tomography shows an air cleft between the esophagus and the left main bronchus (in the red circles), suggesting bronchoesophageal fistula and aspiration pneumonia. (C) Esophagography shows contrast media leakage from the esophagus to the left bronchus (red arrows).

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  Fig. 2. (A, B) Esophagogastroduodenoscopy shows a deep ulcer (in the green circles) in the esophagus at 28 to 32 cm from upper incisor.

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  Fig. 3. (A, B) Esophagogatroduodenoscopic biopsy shows granulomatous inflammation with granulation tissue and marked necrosis (in the red circles). Hematoxylin and eosin stain: (A) ×100, (B) ×200. (C, D) Endobronchial ultrasound with transbronchial needle aspiration shows chronic granulomatous inflammation with necrosis (in the red circle), and many acid-fast positive bacilli (red arrows) are identified by acid-fast bacilli staining. (C) Hematoxylin and eosin stain ×200. (D) Acid-fest bacilli stain ×400.

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  Fig. 4. (A) An esophageal ulcer (green arrows) was seen on esophagogastroduodenoscopy. (B–D) A covered esophageal stent was inserted.

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  Fig. 5. (A, B) Esophagography 2 months after diagnosis shows persistent contrast media leakage from the esophagus to the left bronchus (red arrows). (C, D) Esophagography 9 months after diagnosis shows no contrast media leakage.

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