Yeh Heng, Wu Ren-Chin, Tsai Wen-Sy, Kuo Chia-Jung, Su Ming-Yao, Chiu Cheng-Tang, Le Puo-Hsien
School of Medicine, College of Medicine, Chang Gung University, Taoyuan, Taiwan.
Department of Pathology, Chang Gung Memorial Hospital, Linkou Branch, Taoyuan, Taiwan.
BMC Gastroenterol. 2021 May 8;21(1):206. doi: 10.1186/s12876-021-01801-w.
Systemic lupus erythematosus (SLE) is a multisystemic autoimmune disease, and few cases combine with Crohn's disease. We present the first SLE patient concurrent with Crohn's disease and rectovaginal fistula. She was successfully treated with vedolizumab and surgical intervention. Besides, she also had a rare opportunistic infection, cryptococcal pneumonia, in previous adalimumab treatment course.
A 57 year-old female had SLE in disease remission for 27 years. She suffered from progressive rectal ulcers with anal pain and bloody stool, and Crohn's disease was diagnosed. She received adalimumab, but the lesion still progressed to a rectovaginal fistula. Besides, she suffered from an episode of cryptococcal pneumonia under adalimumab treatment course. Therefore, we changed the biologics to vedolizumab, and arrange a transverse colostomy for stool diversion. She had clinical remission without active inflammation, but the fistula still persisted. Then, she received a restorative proctectomy with colo-anal anastomosis and vaginal repair. Follow-up endoscopy showed no more rectal ulcers or fistula tracts, and contrast enema also noted no residual rectovaginal fistula.
When a SLE patient had unusual rectal ulcers, Crohn's disease should be considered. Biologics combined with surgical intervention is an optimal solution for Crohn's disease with rectovaginal fistula. Although cryptococcal pneumonia is a rare opportunistic infection in the biological treatment, we should always keep it in mind.
系统性红斑狼疮（SLE）是一种多系统自身免疫性疾病，合并克罗恩病的病例较少。我们报告首例并发克罗恩病和直肠阴道瘘的SLE患者。她通过维多珠单抗和手术干预成功治愈。此外，她在之前使用阿达木单抗治疗过程中还发生了罕见的机会性感染——隐球菌肺炎。
一名57岁女性，SLE病情缓解27年。她出现进行性直肠溃疡，伴有肛门疼痛和便血，被诊断为克罗恩病。她接受了阿达木单抗治疗，但病变仍进展为直肠阴道瘘。此外，她在阿达木单抗治疗过程中发生了一次隐球菌肺炎。因此，我们将生物制剂换为维多珠单抗，并安排横结肠造口术以改道粪便。她临床缓解，无活动性炎症，但瘘管仍持续存在。随后，她接受了保留肛门的直肠切除术和结肠肛管吻合术以及阴道修补术。随访内镜检查显示不再有直肠溃疡或瘘管，结肠造影也未发现残留的直肠阴道瘘。
当SLE患者出现不寻常的直肠溃疡时，应考虑克罗恩病。生物制剂联合手术干预是治疗克罗恩病合并直肠阴道瘘的最佳解决方案。尽管隐球菌肺炎是生物治疗中罕见的机会性感染，但我们应始终牢记。
