SEARCH

SEARCH BY CITATION

To the Editor:

Ileal pouch-anal anastomosis (IPAA) has become the surgical procedure of choice in ulcerative colitis (UC) patients who require colectomy. When pouch is constructed, the ileal reservoir is anastomosed with a 2-cm rectal stump or anal transitional zone (ATZ). There are two commonly used anastomotic techniques, the handsewn approach with mucosectomy and the stapled approach without mucosectomy. Inflammation of the rectal cuff or cuffitis is one of the common inflammatory complications of IPAA. It is believed that cuffitis represents a unique form of UC. Here we report a case in which the patient had UC and IPAA and developed de novo collagenous cuffitis with concurrent endoscopic and histologic ischemic changes of cuff mucosa, suggesting ischemia may contribute to development of collagenous cuffitis.

The patient was a 34-year-old Caucasian male who presented with urgency, increased bowel frequency, and pelvic discomfort at our Pouchitis Clinic. He had UC diagnosed 6 years ago and was treated with 5-aminosalicylates, prednisone, azathioprine, infliximab, and adalimumab. He developed medically refractory disease and underwent two-stage stapled J-pouch surgery 2008 at an outside institution. Immediately after ileostomy closure he developed symptoms of urgency, diarrhea, and perianal discomfort. Review of proctocolectomy specimens confirmed the diagnosis of UC (Fig. 1A).

thumbnail image

Figure 1. Histology and endoscopy. (A) Histopathology of proctocolectomy showed diffuse chronic active colitis characterized by cryptitis, crypt abscesses, basal lymphoplasmacytosis, and architectural distortion, features of UC (H&E 100×). There is no evidence of ischemia or collagenous colitis. (B) Pouchoscopy showed a large superficial ulcer of the cuff distal to the anastomosis, occupying 50% of the circumferential area (arrowed area). (C,D) Histology of the anal transitional zone biopsy showed focal fibroplasia in the lamina propria, attenuated crypts, soughing of surface epithelia, and thickened subepithelial collagen band with trapped capillaries (C, H&E stain, 200×), which was confirmed by trichrome stain (D, 200×). [Color figure can be viewed in the online issue, which is available at wileyonlinelibrary.com.]

Download figure to PowerPoint

We did a repeat pouch endoscopy that showed a normal pouch body and afferent limb with ulcers along the suture line. In addition, there was a large superficial ulcer of the ATZ, distal to the anastomosis, occupying 50% of the circumferential area. The endoscopic features were suggestive of ischemia (Fig. 1B). Biopsies of the ulcers in the ATZ showed focal fibroplasia of the lamina propria, attenuated crypts, sloughing of epithelial layer, and collagen deposits beneath the surface epithelium with trapped capillary in the collagen band consistent with collagenous cuffitis on hematoxylin and eosin (H&E) (Fig. 1C) and trichrome (Fig. 1D) stains. In contrast to typical collagenous colitis, collagenous cuffitis in this case showed no evidence of surface epithelial lymphocytosis. After he failed medical therapy with topical mesalamine and corticosteroids, the patient underwent lysis of adhesion and pouch revision surgery, resulting in resolution of collagenous cuffitis.

Cuffitis has been considered a form of UC. Endoscopic features of classic cuffitis were similar to that seen in UC.1 We speculate that cuffitis may share pathogenetic pathways with UC, as patients with typical cuffitis share histologic features of UC, such as crypt distortion, crypt abscess, and neutrophil and mononuclear cell infiltration. However, this case with endoscopic and histologic features of concurrent ischemia and subepithelial collagen deposit without obvious surface epithelial lymphocytosis, its sequential development, and response to surgery may support the notion that cuffitis may represent a spectrum of disease processes with different etiopathogenetic pathways.2

The association between collagenous cuffitis and collagenous colitis is not clear. Several lines of evidence suggest that collagenous colitis be associated with medicines (such as nonsteroidal antiinflammatory drugs3 and lansoprazole4), infections, and an abnormal immune response to luminal antigens.5–8 The endoscopic and histologic features of this case suggest that ischemia may also play a role in the pathogenesis of collagenous colitis or cuffitis.

This is the first reported case of de novo collagenous cuffitis in pouch patients. Cuffitis may represent a spectrum of disease processes. The coexisting ischemic features on endoscopy and histology and good response to lysis of adhesion and pouch revision surgery suggest that ischemia may contribute the development of characteristic collagen band deposits in the gut.

REFERENCES

  1. Top of page
  • 1
    Shen B, Lashner BA, Bennett AE, et al. Treatment of rectal cuff inflammation (cuffitis) in patients with ulcerative colitis following restorative proctocolectomy and ileal pouch-anal anastomosis. Am J Gastroenterol. 2004; 99: 15271531.
  • 2
    Shen B, Lian L, Remzi FH, et al. Natural history of cuffitis in ulcerative colitis patients with restorative proctocolectomy and ileal pouch-anal anastomosis. Poster at Digestive Disease Week 2010, New Orleans, LA.
  • 3
    Kakar S, Pardi DS, Burgart LJ. Colonic ulcers accompanying collagenous colitis: implications of non-steroidal anti-inflammatory drugs. Am J Gastroenterol. 2003; 98: 18341837.
  • 4
    Wilcox GM, Mattia A. Collagenous colitis associated with lansoprozole. J Clin Gastroenterol. 2002; 34: 164166.
  • 5
    Jarnerot G, Tysk C, Bohr J, et al. Collagenous colitis and fecal stream diversion. Gastroenterology. 1995; 109: 449455.
  • 6
    Bohr J, Tysk C, Eriksson S, et al. Collagenous colitis: a retrospective study of clinical presentation and treatment in 163 patients. Gut. 1996; 39: 846851.
  • 7
    Makinen M, Niemela S, Karttunen TJ. Collagenous colitis and Yersinia enterocolitica infection. Dig Dis Sci. 1998; 43: 13411346.
  • 8
    Khan MA, Brunt EM, Longo WE, et al. Persistent Clostridium difficile colitis: a possible etiology for the development of collagenous colitis. Dig Dis Sci. 2000; 45: 9981001.

Bo Shen MD*, Xiuli Liu MD, PhD*, * Departments of Gastroenterology and Anatomic Pathology, Cleveland Clinic Foundation, Cleveland, Ohio.