Clinical Features of Ileal Pouch Polyps in Patients with Underlying Ulcerative Colitis

Purpose Polypoid lesions rarely occur in the ileal pouch in ulcerative colitis patients after restorative proctocolectomy. Clinical features, malignant potential, and management of pouch polyps have not been characterized. Methods We identified 23 ulcerative colitis patients with large polyps (size≥1 cm) of the ileal pouch from our 2,512-case ulcerative colitis pouch database. Demographic, clinical, endoscopic, and histologic data were reviewed. The Pouchitis Disease Activity Index symptom score (range, 0–6) was used to quantify patients’ symptoms before and after polypectomy. Results Of the 23 patients, 95.7 percent (22 patients) had pouch endoscopy indicated for the evaluation of symptoms when polyps were detected, and 60.9 percent of patients had the polyps in the pouch, 26.1 percent in the anal transitional zone, and 21.7 percent in the afferent limb. The mean size of pouch polyps was 1.9 cm ± 1 cm. Twenty-one patients (91.3 percent) had concomitant pouchitis, cuffitis, or Crohn’s disease. On histology, 21 patients (91.3 percent) had inflammatory-type polyps, and 2 (8.7 percent) had dysplastic or malignant polyps. In 18 patients who had endoscopic polypectomy with concurrent medical therapy, the prepolypectomy and postpolypectomy mean symptom scores were 3.4 ± 1.7 and 1.1 ± 1.2 points, respectively (P = 0.015). Two patients (8.7 percent) had pouch excision for malignancy or for concomitant chronic refractory pouchitis. Conclusions The majority of patients with large ileal pouch polyps were symptomatic. These polyps were typically detected on the background of pouchitis, cuffitis, or Crohn’s disease. Although the majority of polyps were inflammatory type, polyps in two patients were dysplastic or malignant. Endoscopic polypectomy with concomitant medical therapy seemed to improve patients’ symptom scores. Supported in part by a NIH grant R03 DK 067275 and an American College of Gastroenterology Clinical Research Award (to B.S.). Poster presentation at meeting of the American College of Gastroenterology, Honolulu, Hawaii, October 30 to November 2, 2005. Reprints are not available.     

Adenocarcinoma in the Anal Canal After Ileal Pouch-Anal Anastomosis for Ulcerative Colitis Using a Double Stapling Technique

A case of adenocarcinoma, developed in the anal canal after ileal pouch-anal anastomosis for ulcerative colitis using a double stapling technique, is reported. In this case a T3N0 cecal cancer was found unexpectedly in the colectomy specimen. Two years later, this patient presented with an outlet obstruction of the pouch because of development of an adenocarcinoma of the anal canal. This was treated with an abdominoperineal excision of the pouch and anorectum.     

Intermittent Torsion of a Megapouch: Report of a Case

We report an unusual presentation of pouch dysfunction because of excessive pouch enlargement or “mega pouch” and probable torsion. The patient presented with abdominal pain that was positional. Contrast-enhanced computerized tomography showed pouch anastomotic staples extending into the right upper quadrant. At operation, gross pouch enlargement with dilation of the afferent ileum was confirmed. Reduction pouch-plasty resulted in pain resolution and maintenance of satisfactory function.     

Adenocarcinoma in an Ileoanal Pouch Formed for Ulcerative Colitis in a Patient With Primary Sclerosing Cholangitis and a Liver Transplant: Report of a Case and Review of the Literature

This is a report of the first case of adenocarcinoma arising in an ileal pouch after proctocolectomy for ulcerative colitis in a patient who had also undergone orthotopic liver transplantation for primary sclerosing cholangitis. Previously reported cases of adenocarcinoma developing after formation of an ileoanal pouch are reviewed as is the evidence for neoplastic transformation of the ileal mucosa. The risk factors for the development of colorectal cancer in patients with ulcerative colitis and the possibility that these may be risk factors for the development of pouch malignancy are discussed. We conclude that this patient exemplifies a small group of patients who may be at increased risk of developing pouch malignancy and need endoscopic follow-up. There is also the need for longer-term follow-up data to determine the risk of this rare and potentially devastating complication of restorative proctocolectomy.     

Infliximab in Ulcerative Colitis is Associated with an Increased Risk of Postoperative Complications After Restorative Proctocolectomy

Purpose  Little data exist regarding infliximab use in surgical decision making and postoperative complications in ulcerative colitis. Our goals were to determine the rate of postoperative complications in infliximab-treated ulcerative colitis patients undergoing restorative proctocolectomy and to determine whether three-stage procedures are more often necessary. Methods  We studied a group of infliximab-treated patients and matched control subjects who underwent two-stage restorative proctocolectomy between 2000 and 2006. Postoperative complications were compared. In addition, the rate of three-stage procedures was compared between all infliximab- and noninfliximab-treated patients. Results  A total of 523 restorative proctocolectomies were performed. In the infliximab group, there were 46 two-stage and 39 three-stage procedures. Covariate-adjusted odds of early complication for the inflixmab group was 3.54 times that of controls (P = 0.004; 95 percent confidence interval (CI), 1.51–8.31). The odds of sepsis were 13.8 times greater (P = 0.011; 95 percent CI, 1.82–105) and the odds of late complication were 2.19 times greater (P = 0.08; 95 percent CI, 0.91–5.28) for infliximab. The odds of requirement for three-stage procedures was 2.07 times greater in the infliximab group (P = 0.011; 95 percent CI, 1.18–3.63). Conclusions  Infliximab increases the risk of postoperative complications after restorative proctocolectomy and has altered the surgical approach to ulcerative colitis. Potential benefits of infliximab should be balanced against these risks. Dr. Sandborn is a consultant (fees paid to Mayo Clinic) to Centocor, Abbott Laboratories, and UCB Pharma; research support for Centocor, Abbott Laboratories, and UCB Pharma. Dr. Pardi is a consultant to Abbott, Salix, Lonza, Elan, and Biobalance; research support for Salix, Proctor & Gamble, Astra Zeneca, Genzyme, Ocera, Optimer Pharmaceuticals, and Massachusetts Biologic Laboratories. Supported by the Edward and Josephine Story Cleveland Clinic Ileal Pouch Database Fund. Read at the meeting of The American Society Colon and Rectal Surgery, St. Louis, Missouri, June 2 to 7, 2007.     

Perineal Adenocarcinoma Thirty Years After Proctocolectomy: Report of a Case

This report describes an adenocarcinoma arising in the perineum 30 years after two-stage total proctocolectomy for ulcerative colitis. This is one of the longest intervals reported between resection and presentation with cancer and also the only case in which the tumor is clearly free of association with existing bowel. The mass originally presented as a perineal mucocele, an unusual complication of proctocolectomy for ulcerative colitis, and this is the first such mucocele to demonstrate malignant transformation. Reprints are not available.     

A Multimodality Approach to Successful Ileal Pouch Desmoid Excision with Pouch Salvage: Report of a Case

Desmoid tumors occur infrequently in patients who undergo proctocolectomy for familial adenomatous polyposis but may result in significant morbidity and mortality depending on the sight of desmoid location. A case of successful ileal pouch salvage using a multimodality approach for treatment of a large ileal pouch associated desmoid tumor is presented. This approach used neoadjuvant chemotherapy to induce a partial response, followed by complete surgical excision with pouch preservation. This is the first reported case of combined chemotherapy and surgical treatment of a desmoid tumor involving an ileal pouch, and the second reported successful attempt at surgical excision with pouch salvage.     

Ulcerative Colitis Accompanied with Sarcoidosis and Dermatomyositis: Report of a Case

We report a rare case of ulcerative colitis with sarcoidosis and dermatomyositis. A 33-year old woman had a ten-year history of ulcerative colitis and dermatomyositis. After nine-year maintenance treatment with sulphasalazine and steroids, she underwent a proctocolectomy and ileal pouch-anal anastomosis because of deterioration of ulcerative colitis. One year after the operation, she noticed some subcutaneous masses in the right forearm and left lower leg. A total biopsy of the mass was performed, and noncaseous epithelioid granulomas were found in the specimen. Chest roentgenogram and chest computed tomography showed hilar adenopathy and a diffuse granular shadow. The diagnosis of sarcoidosis was made. Concomitant development of ulcerative colitis and sarcoidosis may not be incidental, and there may be a common factor in the two diseases. Ulcerative colitis also may be a part of a systemic disorder associated with abnormal immune reactions. Complications by disorders associated with other immunologic abnormalities, as noted in this patient, should be kept in mind in the management of ulcerative colitis. Presented at the Digestive Disease Week (DDW)-Japan 2005 meeting, Kobe, Hyogo, Japan, October 5 to 8, 2005.     

A Case Report: First Case of Filtration Leukocytapheresis for a Patient of Aortitis Syndrome Associated with Ulcerative Colitis

Abstract: An 18‐year‐old woman was treated with leukocytapheresis (LCAP) for her combined ulcerative colitis (UC) and aortitis syndrome (AS). Because a close relationship between these two diseases has been suspected based on their etiological and/or pathological findings, we had hypothesized that LCAP, which has satisfactory effects on inflammatory bowel disease such as UC and Crohn's disease might be effective for both her UC and her AS. After informed consent, LCAP therapy was performed once a week for a total of 7 times. Endoscopic remission of the UC was observed. Even though there were no significant improvements in her subjective symptoms of AS such as side‐neck pain and dizziness, objective evidence of improvement was obtained when the patient's condition was compared before and after LCAP by angiography, angio‐magnetic resonance imaging, and the plethysmogram of her fingertips. These results suggest that LCAP may be valuable as a new adjunct therapy for AS.     

Adenocarcinoma in the anal canal after heal pouch-anal anastomosis for familial adenomatous polyposis using a double-stapled technique: Report of two cases

Restorative proctocolectomy with an ileal pouch-anal anastomosis is thought to abolish the risk of colorectal adenoma development in patients suffering from familial adenomatous polyposis. Both after mucosectomy with a handsewn anastomosis and after a double-stapled anastomosis, rectal mucosa is left behind at the anastomotic site. This carries the potential for the development of polyps and a subsequent malignancy. In our clinic, two patients recently developed an adenocarcinoma at the anastomotic site, despite a yearly follow-up endoscopy.A 40-year-old female underwent an ileal pouch-anal anastomosis with a double-stapled anastomosis in 1991. She refrained from follow-up for several years, but returned eight years postoperatively with a fistula at the anastomotic site. Biopsies revealed an adenocarcinoma infiltrating in the fistula tract T2N0M0. The patient was treated with preoperative radiotherapy (60 Gy), abdominoperineal resection, and a permanent ileostomy.A 27-year-old male underwent an ileal pouch-anal anastomosis with a double-stapled anastomosis in 1990. Because of his profession, endoscopy was performed only once every two years. Endoscopic biopsies ten years postoperatively revealed adenocarcinoma T4N0M0. The patient underwent an abdominoperineal resection with partial resection of the prostate, and a permanent ileostomy was constructed.     

Resolving Microcarcinoids in Ulcerative Colitis: Report of a Case

Ulcerative colitis is known to predispose to the development of neoplasia, especially adenocarcinoma. Microcarcinoids represent small nests of gut endocrine cells located in the mucosa and submucosa of the bowel. Such lesions have been identified in association with chronic inflammation and the concern is that they may represent a precursor lesion for invasive carcinoid tumors. Yet carcinoid tumors are rarely reported in patients with ulcerative colitis. This case report documents a 56-year-old male with ulcerative colitis who was found on random biopsies to have microcarcinoids in his rectal submucosa. Following treatment of his colitis, there was complete resolution of both the inflammation and the microcarcinoids. However, on subsequent follow-up at six months, the patients colitis has returned and so have the microcarcinoids. We explore the issue of whether these lesions represent true neoplasias that should be resected, or whether they represent cellular hyperplasia in response to the inflammatory stimulus.Reprints are not available.     

Septic Complications after Restorative Proctocolectomy do not Impair Functional Outcome: Long-Term Follow-Up from a Specialty Center

Purpose  After restorative proctocolectomy, 7 to 8 percent of patients may have a pouch leak. Concern exists that pouch leak may be associated with impaired functional outcome. We evaluated patients who underwent restorative proctocolectomy to determine whether pouch leak adversely affected long-term functional outcome and quality of life. Methods  We queried our prospectively maintained database of patients who underwent restorative proctocolectomy for demographic and clinical data. We sent a long-term outcome questionnaire to patients, including the validated Fecal Incontinence Severity Index and Cleveland Global Quality of Life scores. Pouch leak was identified by clinical or radiographic evidence of leak. Patients with leak were compared with those without to determine the impact on long-term functional outcome or quality of life. Results  A total of 817 patients were available for follow-up and 374 patients (46 percent) completed questionnaires. The group with (n = 60; 16 percent) and without (n = 314; 84 percent) leak had similar demographics. The median Fecal Incontinence Severity Index score (15.3 vs. 14.7, P = 0.77), Cleveland Global Quality of Life score (0.79 vs. 0.81, P = 0.48), and bowel movements per 24 hours (7.92 vs. 7.88, P = 0.92) were similar. The pouch loss/permanent ileostomy rate was higher in those who leaked (13.3 vs. 0.9 percent, P < 0.001). Conclusions  Anastomotic leak after restorative proctocolectomy does not adversely affect long-term quality of life or functional outcome. However, pouch loss/permanent ileostomy is significantly more likely in patients who have had an anastomotic leak. Read at the meeting of The American Society of Colon and Rectal Surgeons, Boston, Massachusetts, June 7 to 11, 2008. Reprints are not available.     

Prognostic Value of Terminal Ileal Inflammation in Patients with Ulcerative Colitis

Background/AimsFew studies have investigated terminal ileal lesions and their prognostic value in patients with ulcerative colitis (UC). We evaluated the clinical significance of these lesions as a prognostic factor in patients with UC who were in clinical remission.MethodsWe retrospectively selected 567 of 4,066 colonoscopy reports that included positive findings from orificial observations of the terminal ileum (TI) and appendix in patients with UC. We finally recruited patients who were in clinical remission (n=204). We compared the clinical courses, including relapse and other prognostic parameters associated with UC, between the groups.ResultsThe baseline patient characteristics were not significantly different between patients with (n=69, TI+ group) and without TI lesions (n=135, TI– group), although there were more never-smokers in the TI+ group (n=57 [82.6%] in the TI+ group; n=86 [63.7%] in the TI– group; p=0.005). Of note, appendiceal orifice inflammation (AOI) was less frequently found in the TI+ group (14.5%) than in the TI– group (71.9%, p<0.001). The cumulative relapse rate was numerically higher in the TI– group, but it was not significantly different according to the Kaplan-Meier analysis (p=0.116). Multivariate Cox regression analysis also revealed advanced age at diagnosis as the most significant factor (adjusted hazard ratio, 0.964; 95% confidence interval, 0.932 to 0.998; p=0.037), but neither TI inflammation nor AOI were significantly associated with the cumulative relapse rate in patients with UC in clinical remission.ConclusionsFor patients with UC in clinical remission, neither terminal ileal lesions nor AOI had significant clinical or predictive value for future relapse.     

Hemophagocytic Syndrome Caused by Fulminant Ulcerative Colitis and Cytomegalovirus Infection: Report of a Case

We report a case of hemophagocytic syndrome that developed in a 35-year-old, Japanese male with fulminant ulcerative colitis. The patient underwent an emergency operation, consisting of subtotal colectomy, ileostomy with rectal preservation (suprapubic mucous fistula). After the operation, peripheral blood counts showed progressive pancytopenia and bone marrow aspirate smears revealed hypocellular bone marrow with an increase in histiocytes, indicating hemophagocytic syndrome. Viral studies (serum antibody titer and antigenemia of cytomegalovirus) revealed systemic cytomegalovirus infection. The patient was diagnosed with virus-associated hemophagocytic syndrome and was successfully treated with antiviral therapy consisting of intravenous ganciclovir, gamma globulin, and granulocyte-colony stimulating factor.     

Endoscopic Transanal Rectal Mucosal Ablation in the Surgical Treatment of Ulcerative Colitis: Preliminary Results of a Novel Technique

PURPOSE We describe a new technique that endoscopically eradicates rectal stump mucosa after total colectomy for ulcerative colitis.METHODS Seven patients (5 males; median age, 56 (range, 36–72) years) underwent attempted endoscopic transanal rectal mucosal ablation using the 28-French-gauge urologic resectoscope, either at the time of total colectomy and ileostomy for failed medical therapy (5 patients) or as an alternative to completion proctectomy (2 patients) with rectal stump discharge. All had declined restorative proctocolectomy. Clinical, endoscopic, and histologic follow-up was undertaken during a mean of 15 (range, 3–28) months.RESULTS The operative technique evolved during these cases; mucosal ablation was successfully performed leaving a denuded muscular rectal tube in situ in six patients. Mean operative time was 45 minutes. Postoperative endoscopic surveillance has not demonstrated any viable rectal mucosa in these six patients, with only granulation tissue detected histologically. Narrowing of the rectal tube has occurred in two patients. Although all patients report insignificant rectal discharge, urinary and sexual function have remained unchanged.CONCLUSIONS Diathermy ablation of the rectal mucosa via endoscopic transanal rectal mucosal ablation avoids the complications of pelvic dissection and might offer an effective alternative to proctectomy for ulcerative colitis.Presented at the meeting of the Association of Surgeons of Great Britain and Ireland, Manchester, England, May 7 to 9, 2003.     

Squamous-Cell Carcinoma Developing After an Ileoanal Pouch Procedure: Report of a Case

PURPOSE This study was designed to report a new variant of a rare but serious complication of restorative proctocolectomy.METHODS We present a 47-year-old female who underwent restorative proctocolectomy after 16 years of disease. Twenty-five years after her pouch procedure, she underwent pouchoscopy for fever and poor pouch function. A suspicious mass was biopsied and pathology indicated squamous metaplasia. On referral, a mass could be palpated above the anorectal ring. Biopsy of the mass was read as invasive squamous carcinoma in the background of normal intestinal mucosa. This represents the twelfth reported case of carcinoma arising in a pouch, but the first report of a squamous carcinoma, as all previous reports had been of adenocarcinoma.RESULTS The patient has undergone chemoradiation. Response to therapy, functional status, and biopsy after treatment will determine whether the patient will be able to salvage the pouch.CONCLUSIONS Diligence and vigilance with regard to active follow-up, and a high index of suspicion, are required to prevent this from becoming a more frequently seen problem.Reprints are not available.