Introduction and importance: Gastrointestinal (GI) hemorrhage due to arterio-enteric fistula (AEF) is exceedingly rare. In adults, reported cases are often secondary to prior vascular surgery or underlying vasculitic disorders. In children, reports of secondary AEFs are scarce, and instances of primary AEF are exceptionally rare. Case presentation: A 17-month-old boy was admitted with 1 day of melena. On examination, he was pale and tachycardic, with a soft abdomen. The hemoglobin was 6.3 g/dl, with a normal coagulation profile and platelet count. After transfusion of 10 ml/kg packed red blood cells, the patient was stabilized with hemoglobin rising to 9.5 g/dl. Computed tomography (CT) scan and GI endoscopy were scheduled to investigate the source of bleeding. CT scan revealed a 9-mm right external iliac artery aneurysm without contrast extravasation into the intestinal tract. On day 3, the patient had massive hematochezia with shock. Emergency GI endoscopy showed no clear bleeding source. Exploratory laparotomy revealed an inflamed right external iliac artery aneurysm with fistulization into the jejunum. The aneurysm was resected with vascular repair, and the affected jejunal segment was resected with anastomosis. Postoperatively, the patient had no further bleeding, recovered gradually, and was discharged on day 13. At 1-month follow-up, he remained asymptomatic; Doppler ultrasound showed an intact right iliac artery without thrombosis or recurrent aneurysm. Conclusion: AEF in children is exceedingly rare and usually presents with severe acute GI bleeding. A CT scan can detect vascular abnormalities and suggest the diagnosis. Prompt surgical management combined with endoscopy is the appropriate life-saving therapeutic approach.
Tran et al. (Tue,) studied this question.