Kanaka Shintaro, Yamada Takeshi, Matsuda Akihisa, Takahashi Goro, Hara Keisuke, Yoshida Hiroshi
Department of Gastrointestinal and Hepato-Biliary-Pancreatic Surgery, Nippon Medical School, Tokyo, Japan.
J Anus Rectum Colon. 2020 Oct 29;4(4):201-205. doi: 10.23922/jarc.2020-029. eCollection 2020.
Vascular Ehlers-Danlos syndrome (vEDS) is a rare autosomal dominant connective tissue disease. Patients with vEDS are at a high risk of developing severe complications (such as arterial aneurysm, arterial rupture, intestinal rupture) at an early age. We report a case of colonic perforation in a vEDS patient with no family history of that disease. A 28-year-old man with abdominal pain arrived at our hospital in an ambulance. The preoperative diagnosis was panperitonitis due to gastrointestinal perforation. Although his parents had not suffered from vEDS, he had been diagnosed with the disease at 25 years of age because of his history of arterial dissection. We performed an emergency operation using Hartmann's procedure to construct a descending colostomy. There remains a lack of consensus on surgical management in vEDS patients with gastrointestinal perforation because of the limited number of reported cases.
血管型埃勒斯-当洛综合征(vEDS)是一种罕见的常染色体显性遗传性结缔组织疾病。vEDS患者在早年发生严重并发症(如动脉瘤、动脉破裂、肠破裂)的风险很高。我们报告一例无该疾病家族史的vEDS患者发生结肠穿孔的病例。一名28岁腹痛男性乘救护车抵达我院。术前诊断为胃肠穿孔所致的全腹膜炎。尽管他的父母未患vEDS,但他因有动脉夹层病史,在25岁时被诊断患有该疾病。我们采用哈特曼手术进行急诊手术,行降结肠造口术。由于报道的病例数量有限,对于vEDS合并胃肠穿孔患者的手术治疗仍缺乏共识。