Abstract
BACKGROUND: Adenoid cystic carcinoma (ACC) is a rare malignant tumor often involving the central airways. Airway stenosis is a common complication in these patients especially following surgery and radiotherapy. Airway stenting provides effective palliation in patients unsuitable for surgical reintervention. However, serious complications such as bronchoesophageal fistula (BEF) remain rare and underrecognized. CASE PRESENTATION: We reported the first documented case of BEF occurring after airway stenting for ACC-related airway stenosis. The patient had a 13-year history of recurrent ACC involving the right main bronchus, previously treated with surgery, radiotherapy, and multiple bronchoscopic interventions. Two Y-shaped metallic stents were sequentially placed and later replaced with a Y-shaped silicone stent to facilitate removability and reduce granulation formation. Although airway patency was initially maintained, the patient subsequently developed nonspecific symptoms of worsening cough and sputum retention. The fistula was identified only after silicone stent removal, when bronchoscopy revealed a defect in the left main bronchus that was confirmed on contrast-enhanced CT. CONCLUSIONS: BEF following airway stenting for ACC is a rare but serious complication. Clinicians should exercise caution in managing stents in patients with irradiated, cachectic, and recurrent ACC, balancing theoretical device advantages against airway fragility and systemic vulnerability. Individualized stent selection, pre-removal contrast-enhanced CT, optimization of nutritional and infectious status, and avoidance of unnecessary stent exchanges are essential for reducing the risk of this life-threatening complication.