Shams Christienne, Hakim Seifeldin, Amin Mitual, Cappell Mitchell S
Division of Gastroenterology & Hepatology, Department of Medicine, William Beaumont Hospital, 3535 W. Thirteen Mile Rd, Royal Oak, MI 48073, USA.
Department of Pathology, William Beaumont Hospital and Oakland University William Beaumont School of Medicine, 3601 W Thirteen Mile Rd, Royal Oak, MI 48073, USA.
Case Rep Gastrointest Med. 2018 Jul 30;2018:7506069. doi: 10.1155/2018/7506069. eCollection 2018.
Prepouch ileitis (PI) is an uncommon complication of ileal pouch anal anastomosis (IPAA) and restorative proctocolectomy (RPC) for treatment of refractory ulcerative colitis (UC). A case is reported of PI in a 16-year-old girl who presented with severe UC that was initially stabilized with infliximab therapy but re-presented 1 year later with severe UC, refractory to infliximab and corticosteroid therapy, which required IPAA and RPC. Her symptoms resolved postoperatively, but she re-presented 1 year later with 10 loose, bloody, bowel movements/day and involuntary 6-Kg weight-loss. Computerized tomographic enterography showed focal narrowing and mucosal enhancement of the pouch and focal narrowing, abnormal mucosal enhancement, and mural thickening of the prepouch ileum. Pouchoscopy revealed exudates and ulcerations in both the pouch and prepouch ileum up to 50 cm proximal to pouch, as confirmed by histopathology of pouch and ileal biopsies. Capsule endoscopy revealed no small intestinal lesions beyond 50 cm from the pouch. She required antibiotics, hydrocortisone enemas, and eventually azathioprine to control her symptoms. She remains asymptomatic 4 years later while chronically administered azathioprine therapy. Comprehensive literature review demonstrates that this case illustrates the classical clinical, radiologic, endoscopic, and histopathologic findings in PI, a relatively rare syndrome.
贮袋性回肠炎(PI)是回肠贮袋肛管吻合术(IPAA)和全直肠系膜切除回肠肛管吻合术(RPC)治疗难治性溃疡性结肠炎(UC)时罕见的并发症。本文报道了1例16岁患严重UC的女孩,其病情最初通过英夫利昔单抗治疗得以稳定,但1年后复发,对英夫利昔单抗和皮质类固醇治疗无效,遂行IPAA和RPC。术后其症状缓解,但1年后再次出现症状,每天有10次稀便、便血,体重非自愿减轻6千克。计算机断层扫描小肠造影显示贮袋局部狭窄和黏膜强化,贮袋前回肠局部狭窄、黏膜强化异常及肠壁增厚。贮袋镜检查发现贮袋及贮袋前回肠(距贮袋近端达50厘米)有渗出物和溃疡,贮袋和回肠活检的组织病理学检查证实了这一点。胶囊内镜检查显示距贮袋50厘米以外的小肠无病变。她需要使用抗生素、氢化可的松灌肠剂,最终使用硫唑嘌呤来控制症状。在长期接受硫唑嘌呤治疗4年后,她仍无症状。全面的文献综述表明,该病例说明了PI这一相对罕见综合征的典型临床、放射学、内镜和组织病理学表现。