Esophagobronchial fistula in a patient with Behcet's disease: Report of a case

Esophageal involvement in Behcet's disease is generally considered to be very uncommon. So far, six cases of esophageal ulcers associated with perforation, penetration, or fistula in Behcet's disease have been described in the English literature. This report describes esophagobronchial fistula in a patient with intestinal Behcet's disease. A 62-year-old man was transferred to our hospital for peritonitis due to a small intestinal perforation after an appendectomy. At the age of 14 years he had had recurrent oral ulcers. Ulcerations of the ileum and epididymitis were found, and a pathological examination revealed nonspecific inflammation. Furthermore, an esophageal ulcer with esophagobronchial fistula was diagnosed. The fistula required not only endoscopic treatment but also surgical intervention. The patient's clinical features were consistent with the active phase of intestinal Behcet's disease. The symptoms gradually resolved without any treatment. Four years after remission, however, the symptoms recurred with gastrointestinal hemorrhage and polyarthritis. In the ileocolic region, punch-out ulcerations were noted. The clinical history and features led to a diagnosis of Behcet's disease associated with recurrent gastrointestinal ulcerations. Steroid therapy (prednisolone, 20 mg daily) was started, and led to a rapid resolution of the symptoms. The patient is now being followed up as an outpatient while receiving prednisolone (10 mg per day), without complaint of any gastrointestinal symptoms.