Nakajima Hideaki, Nouso Hiroshi, Urushihara Naoto, Fukumoto Koji, Yamoto Masaya, Miyake Hiromu, Sekioka Akinori, Nomura Akiyoshi, Yamada Yutaka
Department of Pediatric Surgery, Shizuoka Children's Hospital, 860 Urushiyama, Aoi-ku, Shizuoka 420-8660, Shizuoka, Japan.
Case Rep Gastrointest Med. 2018 Nov 25;2018:8087659. doi: 10.1155/2018/8087659. eCollection 2018.
Blue rubber bleb nevus syndrome (BRBNS) is a rare disease in which venous malformations (VMs) involve any body organ, most commonly the skin and the gastrointestinal (GI) tract. Treatment of BRBNS aims at preserving the GI tract as much as possible. Although there are several dozen case reports about BRBNS that describe short-term clinical courses, a few provide an account of long-term clinical course. Here, we report a case of BRBNS in a girl that required multiple abdominal surgeries due to the GI VMs and a recurrence at an interval of 14 years. The preferred approach for gastrointestinal VMs involves the complete resection of all lesions without residual VMs. It is important to bear in mind the possibility of delayed recurrence of GI VMs after surgical or endoscopic treatment.
蓝色橡皮疱痣综合征(BRBNS)是一种罕见疾病,其中静脉畸形(VMs)累及身体任何器官,最常见于皮肤和胃肠道(GI)。BRBNS的治疗旨在尽可能保留胃肠道。虽然有几十篇关于BRBNS的病例报告描述了短期临床病程,但很少有报告长期临床病程。在此,我们报告一例BRBNS女童病例,该患儿因胃肠道VMs需要多次腹部手术,且间隔14年复发。胃肠道VMs的首选治疗方法是完全切除所有病变且无残留VMs。重要的是要记住手术或内镜治疗后胃肠道VMs可能会延迟复发。