Successful use of infliximab for treating fistulizing pouchitis with severe extraintestinal manifestation: A case report

To the Editor:

Surgery is not a nightmare for patients with ulcerative colitis (UC) since 1978, when Park and Nichols first introduced their surgical procedure that restores continuity of the bowel after total proctocolectomy and prevents stoma formation.¹ Ileoanal pouch is the most frequent operation for patients with UC in the absence of contraindications. Anastomotic leak, pouch failure, pelvic abscess, and fistulas originating from the pouch can occur after the procedure but the most common complication is pouchitis, the inflammation of the reservoir. The prevalence of pouchitis ranges from 10%–45%; the frequency of refractory pouchitis is 4.5%–5.5%, and severe form, in which case pouch excision can become necessary, occurring in 0.5%–1% of the patients.² Pouchitis is characterized by increased stool frequency, bloody stool, fecal urgency, incontinence, abdominal or pelvic discomfort, malaise, and fever. We report a case where infliximab induced a rapid remission and effected a long-lasting symptom-free period in a young girl with severe refractory pouchitis associated with pyoderma gangrenosum.

The severe pancolitis of the young girl started in September 2003 at the age of 14, and 2 years later because of steroid dependence, azathioprine- and cyclosporine-resistant chronic activity, ileal-pouch anal anastomosis (IPAA) was performed. She was asymptomatic until the last step of the operation. Within 4 weeks after the closure of the temporary ileostomy, severe pouchitis developed with profuse bloody diarrhea, weakness, anorexia, fever, and significant weight loss. Due to the insufficient response to the standard treatment and the subsequently occurring pelvic abscess and perineal fistula, a temporary ileum deviation had to be performed, again resulting in a complete asymptomatic condition for months. Six weeks after the repeated reconstruction of the bowel continuity, severe pouchitis (Pouchitis Disease Activity Index [PDAI] = 17, worst score is 18)³ set in again, which was accompanied by pyoderma gangrenosum, and indicated infliximab treatment due to the unsuccessful combined antibiotics, topical budesonide, oral corticosteroid, and azathioprine therapy. After the induction with infliximab, including 3 infusions with a dosage of 5 mg/kg, all of the patient's symptoms and complaints ceased, pyoderma gangrenosum healed, IBDQL, PDAI, and the endoscopic findings also demonstrated complete regression (Figs. 1, 2.). Now the patient is on continuous infliximab therapy with 8-weekly regular retreatments for more than 16 months without any flare-up. She restarted her handball career in the first professional league.

Pouchitis is an idiopathic inflammatory condition of the ileal reservoir in patients after proctocolectomy and IPAA with a frequency of up to 60% in cases operated on for UC.² This frequency is significantly higher than patients with familiar adenomatous polyposis, suggesting a permanent pathological immune dysbalance, in fact, of colectomy. Although most patients have a good response to antibiotic therapy, there are a few difficult cases when other therapeutic options (topical and/or oral mesalamine, topical and/or oral steroids, and immunosuppressive treatment) have to be used, while in every fifth case the unsuccessful therapeutic response leads to the removal of the reservoir.

There is little information, some case reports, about the beneficial effect of TNF-α blockage in pouchitis, although the next logical therapeutic step is the use of biologics in the refractory patients. According to our case, anti-TNF-α therapy may be used to induce and maintain remission in chronic, severe pouchitis and in its extraintestinal manifestations nonresponding to other treatment, although the true therapeutic efficiency and safety should be established exactly after further studies.

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