A Case of BRBNS in a Patient who Presented With Intestinal Obstruction and Perforation Following DBE Coagulation Therapy

Abstract

Blue rubber bleb nevus syndrome (BRBNS) is a systemic disease of multiple venous malformations in the gastrointestinal (GI) tract and skin, which causes GI hemorrhage and subsequent anemia. The anemia can be treated conservatively, and endoscopic argon plasma coagulation (APC) can be carried out for GI tract lesions. Sometimes, surgical intervention for BRBNS is indicated to control bleeding, intussusception, ileus, or intestinal ischemia. A 5-year-old girl with BRBNS underwent double-balloon endoscopy (DBE) for the control of GI hemorrhage. On the following day, she experienced abdominal obstruction caused by a large hematoma in the small intestine. Despite conservative treatment for 10 days, the obstruction did not improve, and eventually, intestinal perforation occurred. Emergency laparotomy was carried out, and two lesions of the small intestine were resected. In BRBNS, DBE allows the detection and treatment of gastrointestinal lesions by APC; however, careful follow-up for unexpected complications, such as hematoma, is necessary.