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Duodenal leiomyoma represents a very unusual cause for acute gastrointestinal bleeding. The authors report the case of a 49-year-old male with a massive bleeding from an ulcerated duodenal leiomyoma involving the ampulla of Vater. An emergency Whipple's pancreaticoduodenectomy was carried out under suspicion of local malignancy. It is emphasized that the proper diagnosis of smooth muscle tumors is often difficult, both clinically and from the pathological point of view.
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