Sato Hitoshi, Shima Kentaro, Sakata Hanae, Ohtoh Takashi
Internal Medicine, South Miyagi Medical Center, Ogawara, Japan.
Surgery, South Miyagi Medical Center, Ogawara, Japan.
BMJ Case Rep. 2019 Aug 10;12(8):e230355. doi: 10.1136/bcr-2019-230355.
Intestinal perforation is a rare and life-threatening complication in granulomatosis with polyangiitis (GPA). A 55-year-old Japanese man who had been undergoing maintenance haemodialysis was diagnosed with GPA via a pathologically proven nasal granuloma and proteinase 3-antineutrophil cytoplasmic antibody-positive status. He was started on 60 mg prednisolone. Haematochezia was observed due to the colon ulcer after treatment initiation. Two doses of intravenous cyclophosphamide were administered every 2 weeks. Despite the treatment, there were two episodes of severe abdominal pain with peritonitis within a week. Perforation of descending colon and severe ischaemia of the ascending colon was observed during each emergency laparotomy, and ileostomy was performed. Vasculitis in the small vessels was confirmed. In conclusion, patient with severe intestinal involvement and who was unresponsive to conventional therapy was treated with surgery followed by rituximab administration, and remission was achieved.
肠道穿孔是肉芽肿性多血管炎(GPA)中一种罕见且危及生命的并发症。一名55岁正在接受维持性血液透析的日本男性,经病理证实的鼻肉芽肿和蛋白酶3-抗中性粒细胞胞浆抗体阳性确诊为GPA。开始给予60mg泼尼松龙治疗。治疗开始后因结肠溃疡出现便血。每2周静脉注射2剂环磷酰胺。尽管进行了治疗,但一周内仍有两次严重腹痛伴腹膜炎发作。每次急诊剖腹手术时均观察到降结肠穿孔和升结肠严重缺血,并进行了回肠造口术。证实存在小血管血管炎。总之,对于严重肠道受累且对传统治疗无反应的患者,先进行手术,随后给予利妥昔单抗治疗,实现了缓解。