An Adult Case of Congenital Bronchoesophageal Fistula

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Although bronchoesophageal fistula (BEF) is well-known as a congenital disease, it is rarely seen in adults. Most common causes of acquired BEF in adult are malignancy, infection, trauma, and inflammation. We experienced a case of acquired and idiopathic BEF in an adult, therefore described our rare case and review of manuscripts associated with BEF.A 47-year-old man was admitted to our emergency department with complaints of fever, cough, and dysphagia with liquid ingestion. Information from the hospital from which he was transported indicated that esophageal tissue biopsy disclosed no evidence of malignancy, cytomegalovirus (CMV), or herpes simplex virus. Tuberculosis was not identified by interferon-gamma release assays test and polymerase chain reaction from the sputum. Findings from his general examination on arrival were as follows; heart rate 95 beats per minutes, blood pressure 97/66 mmHg, respiratory rate 16 breaths per minute, oxygen saturation 100% with 2L oxygen, body temperature 36.60 C, Glasgow Coma Scale score E4V5M6. Laboratory examination results were as follows: white blood cells 8700/μ, hemoglobin 10.4g/dl, C-reactive protein 14.3mg/dl.

 Anti-human immunodeficiency virus (HIV) test was negative. His chest radiography showed an abnormal shadow in the left lower field, suggesting pneumonia (Figure 1A). Chest computed tomography revealed a fistula running from the middle esophagus to the left lower bronchus, with pneumonia and atelectasis in the left lower lobe (Figure 1B). Contrast radiography of the upper gastrointestinal tract showed a barium outline of the esophagus, fistulous tract, and the left lower lobe bronchus (Figure 1C). To detect the cause of the fistula, gastrointestinal fiberscopy (GIF) was performed. Multiple longitudinal ulcers and fistulas were recognized (Figure 1D). No malignant or inflammatory tissues were recognized from BEF is well-known as a congenital disease and is thought to have been first reported by Negus in 1929. Frequent causes of acquired BEF include malignancy; on the other hand, benign causes such as tuberculosis, inflammatory conditions like Crohn’s disease, and traumatic factors have been found to be responsible for only 5-6% of cases.

 BEF is divided into fourtypes along with Braimbridge and Keith’s classification of BEF in 1965: type I, esophageal diverticulum with a large ostium and fistula at its tip; type II, a short tract running directly from the esophagus to the trachea or bronchus; type III, a fistulous tract connecting the esophagus with a cyst in the lobe, which also communicates with the bronchus; and type IV, a fistula that leads into a sequestrated lobe or segment. Common complaints are recurrent bouts of cough, hemoptysis, dysphagia, and fever. Esophageal cancer is the major cause of BEF in adults. BEF develops in 5% to 15% of patients with esophageal cancer, leading to life-threatening complications. Treatment and adequate management of BEF is challenging. Kimura et al. reported a patient with advanced esophageal cancer with tracheobronchial fistula treated with esophageal bypass surgery. Fukuhara et al. reported four cases of esophagobronchial fistula treated with stenting [5]. BEF caused by esophageal cancer is a sign of poor prognosis. Palliative treatment and quality of life are the main concerns for these patients.