Fujikawa Hiroyuki, Araki Toshimitsu, Hashimoto Kiyoshi, Okita Yoshiki, Tanaka Koji, Hiro Junichiro, Inoue Yasuhiro, Mohri Yasuhiko, Uchida Keiichi, Kusunoki Masato
Department of Gastrointestinal and Pediatric Surgery, Division of Reparative Medicine, Institute of Life Sciences, Mie University Graduate School of Medicine, Tsu, Mie, Japan.
Int Surg. 2014 Nov-Dec;99(6):705-9. doi: 10.9738/INTSURG-D-13-00084.1.
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.
一名52岁女性被诊断为直肠血管瘤并伴有慢性出血。通过临床检查诊断为克-特综合征。30年后，她因进行性贫血前来就诊。结肠镜检查发现多个蓝色息肉样结节，伴有严重的血管充血和静脉曲张，从齿状线沿肠壁周向蔓延至直肠乙状结肠病变处。选择性腹部血管造影显示血管瘤由乙状结肠动脉和直肠上动脉供血。经直肠多普勒超声检查显示病变不均匀，低回声区主要位于黏膜下层，部分在齿状线至1厘米处的括约肌间层形成彩色血流信号的镶嵌模式。行前切除术及结肠肛管吻合术并覆盖回肠造口术。采用经肛门入路，根据经直肠多普勒超声检查结果进行黏膜切除术和括约肌间切除术。术中无并发症，术后贫血得到缓解。
