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A male patient with an arterio-portal fistula resulting from a mesenteric arteriovenous malformation, who developed portal hypertension and liver cirrhosis, is presented herein. The malformation was considered to be congenital in origin and its location made any ablative surgical procedure impossible. Such alternative treatments as ligation of the afferent arteries, followed by transarterial embolization were therefore given, but both were unsuccessful. We also present a review of the literatures of mesenteric arteriovenous fistula. Radical surgical approach for this rare entity is proposed. The case reported here as related to mesenteric arteriovenous communications of congenital origin is the seventh such case published, and the first which was ever found to be located in the trunk of the superior mesenteric artery.
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