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Polypoid or tumorous arteriovenous malformation (AVM) of small intestine is rare and can be misdiagnosed as other tumorous conditions. We experienced a rare case of giant jejunal AVM in a 15-year-old boy, who complained of intense abdominal pain. Ultrasonography and contrast-enhanced CT revealed a 13.5-cm-sized multiseptated cystic mass arising in small intestine, which was mimicking submucosal tumor. It was successfully treated by surgical resection. The specimen showed a multilobulated outbulging submucosal mass in jejunum. Histopathologic evalulation confirmed AVM located in the submucosa, muscularis propria and subserosa. This case is the largest AVM of small intestine among which has ever been reported.

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