A 68-year-old woman was referred to our hospital for evaluation of severe anemia. Although she had an upper endoscopy and colonoscopy that failed to show the cause of her anemia, there was a small amount of blood in the terminal ileum, which suggested small bowel bleeding. In our hospital, video capsule endoscopy identified a spherically raised lesion in the jejunum, with central erosion of the surface mucosa, which was similar in morphology to a submucosal tumor (Fig. 1a). Balloon-assisted enteroscopy revealed a dark reddish, elevated lesion in the jejunum, the surface of which was extensively peeled off and thickly covered by blood clots (Fig. 1b). Malignant tumors such as angiosarcoma, gastrointestinal stromal tumor, and malignant lymphoma were suspected. A definitive preoperative diagnosis was difficult because of the unusual endoscopic findings. The patient underwent laparoscopic small bowel resection. The lesion was 17 mm in diameter (Fig. 2a). Histological findings revealed that the elevated lesion consisted of exposed vascular components, surrounded by normal small bowel mucosa, and numerous dilatated vessels without atypia were observed from the lamina propria to the muscularis propria. These findings were consistent with cavernous hemangioma (Fig. 2b,c). After surgery, the anemia improved within 6 months. Hemangiomas occur rarely in the small bowel. Thirteen cases of small bowel hemangiomas, observed preoperatively by enteroscopy, have been reported in adults. To the best of our knowledge, a submucosal tumor-like hemangioma with exposed vascular components is extremely rare and has been described in only one case. It was hypothesized that this uncommon morphology was formed by collapse of the tumor due to external stimuli such as small bowel peristalsis and food residue. This case highlights the unusual endoscopic appearance of small bowel hemangioma as a rare cause of obscure gastrointestinal bleeding.
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