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Case Reports
. 2018 Jul 30:2018:7506069.
doi: 10.1155/2018/7506069. eCollection 2018.

Case Report and Literature Review Illustrating the Clinical, Endoscopic, Radiologic, and Histopathologic Findings with Prepouch Ileitis after IPAA and Restorative Proctocolectomy for Refractory Ulcerative Colitis

Christienne Shams  1 , Seifeldin Hakim  1 , Mitual Amin  2 , Mitchell S Cappell  3
Affiliations
Case Reports

Case Report and Literature Review Illustrating the Clinical, Endoscopic, Radiologic, and Histopathologic Findings with Prepouch Ileitis after IPAA and Restorative Proctocolectomy for Refractory Ulcerative Colitis

Christienne Shams et al. Case Rep Gastrointest Med. .

Abstract

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.

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Figures

Figure 1
Figure 1
Colonoscopy with intubation of terminal ileum shows diffusely erythematous and granular mucosa with focal exudation affecting the rectum through ascending colon, with sparing of the cecum and terminal ileum. The endoscopic findings are consistent with UC.
Figure 2
Figure 2
Photomicrograph of histopathology with H&E stain of biopsy specimen shows normal ileal mucosa prior to postoperative occurrence of prepouch ileitis, with relatively normal villous height and preserved villous-to-crypt ratio.
Figure 3
Figure 3
(a) Coronal section of computerized tomographic enterography shows focal mural thickening and enhancement of afferent limb of the J-pouch 1 year after undergoing IPAA and RPC for ulcerative colitis refractory to medical therapy. (b) Transverse section of computerized tomographic enterography shows mucosal hyperenhancement, mural thickening, and luminal narrowing of a 9.4 cm long segment of terminal ileum 1 year after undergoing IPAA and RPC for ulcerative colitis refractory to medical therapy.
Figure 4
Figure 4
(a) Pouchoscopy shows ulceration and mild inflammation in the J-pouch. (b) Pouchoscopy shows ulceration and mild inflammation in the terminal ileum proximal to the J-pouch.
Figure 5
Figure 5
Histopathology with H&E shows near total loss of villi, crypt architectural distortion, and crypt abscesses (arrows) in endoscopic biopsies obtained 20 cm from anal verge, findings highly consistent with prepouch ileitis after creation of a J-pouch.
See this image and copyright information in PMC

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