A 52-year-old woman was diagnosed with rectal hemangioma that had resulted in chronic bleeding. Klippel-Trenaunay syndrome was diagnosed by clinical examination. She was referred 30 years later because of progressive anemia. Colonoscopy revealed multiple bluish, polypoid nodules with severe vascular congestion and varicosis that had circumferentially spread along the wall from the dentate line to the rectosigmoid lesion. Selective abdominal angiography indicated that the hemangioma fed from the sigmoid artery and superior rectal artery. Doppler transrectal ultrasonography showed heterogeneous lesions with hypoechoic areas mostly in the submucosal layer, partly forming the mosaic pattern of the color flow signal in the intersphincteric layer on the oral side from the dentate line to 1 cm. Anterior resection and coloanal anastomosis with covering ileostomy was performed. Using a transanal approach, mucosectomy and intersphincteric resection were performed based on the Doppler transrectal ultrasonography results. There were no intraoperative complications, and her anemia resolved postoperatively.
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