Sasaki K, Nakagawa H, Takahashi T, Sato E
Department of Internal Medicine, Shin-Shirakawa Central Hospital, Shirakawa, Japan.
Am J Gastroenterol. 1991 Jan;86(1):105-8.
An 8-yr-old Japanese boy was hospitalized, complaining of active hematochezia. He was shown to have a cherry-red, blood-oozing area of vascular dilation with mucosal prominence in the sigmoid colon by endoscopy, but had no other mucocutaneous vascular abnormalities. A family history was noncontributory. Under the clinical diagnosis of angiodysplasia of the sigmoid colon, he underwent an elliptical resection of the part, based on the intraoperative endoscopic findings. However, histological examination of the surgical specimen revealed a totally different picture from that seen in angiodysplasia: the thin-walled, markedly ectatic, nontortuous veins with hemorrhage were seen only just below the muscularis mucosae, around which the normal ones were shown to coexist. He has had no rebleeding for the past 3 yr. This lesion is considered to be an isolated congenital visceral telangiectasia involving veins.
一名8岁日本男孩因出现活动性便血入院。内镜检查显示，其乙状结肠有一个樱桃红色、渗血的血管扩张区域，伴有黏膜隆起，但无其他黏膜皮肤血管异常。家族史无特殊。在临床诊断为乙状结肠血管发育异常后，根据术中内镜检查结果，对该部位进行了椭圆形切除。然而，手术标本的组织学检查显示，其情况与血管发育异常完全不同：仅在黏膜肌层下方可见薄壁、明显扩张、无迂曲且伴有出血的静脉，周围有正常静脉并存。在过去3年里，他没有再次出血。这种病变被认为是一种孤立的先天性内脏静脉性毛细血管扩张症。
