Arteriovenous malformation that caused prolapse of the colon and was treated surgically in an infant: a case report
Arteriovenous malformation that caused prolapse of the colon and was treated surgically in an infant: a case report
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DOI：
10.1186/s40792-020-00824-x
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发表时间：
2020-04-08
影响因子：
0.8
通讯作者：
Kohno, Miyuki
中科院分区：
文献类型：
--
作者：
Kido, Miori;Nakamura, Kiyokuni;Kohno, Miyuki
研究背景肠道血管病变有多种描述，包括血管发育不良、动静脉畸形和毛细血管扩张。这种病变在成人中很常见，以血管发育不良（动静脉畸形的一种）为典型。相反，这些病变在儿科人群中很少见。血管发育不良可能导致胃肠道出血，这有时是治疗的指征。考虑到手术治疗后复发率高，主要选择保守治疗。我们在此报告一个极为罕见的情况下，脱垂的结肠由于动静脉畸形成功地切除治疗一个1岁的女孩。我们还强调了儿童和成人病例之间的差异。病例介绍一个女孩在7个月大时出现血便。她在1岁时因持续中度便血和最近发生直肠脱垂到另一家医院就诊。结肠镜检查显示一个位于肛门边缘15厘米处的突出病变，提示粘膜下血管异常。在我院进行的对比增强计算机断层扫描和磁共振成像显示，局部病变伴部分乙状结肠血管扩张;胃肠道中不存在其他病变。行腹腔镜辅助乙状结肠切除术。观察浆膜下血管病变，并使用端端吻合术切除。2.2 x 2.7 cm节段的病理学检查显示粘膜下层中有几条异常增大和扩张的血管延伸至浆膜下。病变被诊断为动静脉畸形。在2年随访时，患者的临床病程良好，无复发。结论乙状结肠动静脉畸形很少引起肠套叠和结肠脱垂。完全切除是一种根治性和潜在有效的治疗方法。计算机断层扫描和结肠镜检查是有用的评估病变在本案件。
Background Various terms have been used to describe vascular lesions in the intestine, including angiodysplasia, arteriovenous malformation, and telangiectasia. Such lesions are common in adults and are typified by angiodysplasia, a type of arteriovenous malformation. In contrast, these lesions are rarely seen in the pediatric population. Angiodysplasia may cause gastrointestinal bleeding, which is sometimes an indication for treatment. Considering the high rate of recurrence after surgical treatment, conservative treatments are mainly chosen. We herein report an extremely rare case of a prolapsed colon due to an arteriovenous malformation successfully treated by resection in a 1-year-old girl. We also highlight the differences between pediatric and adult cases. Case presentation A girl developed bloody stools at 7 months of age. She visited another hospital at 1 year of age because of continuing moderate hematochezia and recent onset of rectal prolapse. Colonoscopy showed a protruding lesion located 15 cm from the anal verge, suggesting a submucosal vascular abnormality. Contrast-enhanced computed tomography and magnetic resonance imaging at our hospital revealed the localized lesion with dilated blood vessels in part of the sigmoid colon; no other lesions were present in the gastrointestinal tract. Laparoscopic-assisted sigmoidectomy was performed. A subserosal vascular lesion was visualized and resected using end-to-end anastomosis. Pathologic examination of the 2.2 x 2.7-cm segment revealed several abnormally enlarged and ectatic blood vessels in the submucosa extending into the subserosa. The lesion was diagnosed as an arteriovenous malformation. The patient had a good clinical course without recurrence at the 2-year follow-up. Conclusions An arteriovenous malformation in the sigmoid colon may rarely cause intussusception and prolapse of the colon. Complete resection is a radical and potentially effective treatment. Computed tomography and colonoscopy were useful for evaluation of the lesion in the present case.