Background Intestinal involvement is one of the intractable categories of Behçet's disease (BD) and closely related with prognosis of patients with BD. Although high dose corticosteroids (CS) and various immunosuppressant are used for treatment, refractory intestinal-BD is difficult to manage, and effective treatment remains elusive. The presence and role of TNF-α in ulcerative lesion of the intestine has been reported, and several case reports including our previous study 1) have shown favorable effects for infliximab (IFX) in patients with refractory intestinal-BD.
Objectives In this study, we evaluate the efficacy and safety of 3-year treatment of IFX in 21 patients with intestinal-BD who were 1) refractory to conventional therapies or 2) difficult to continue treatment because of adverse effects, or 3) not able to taper the dose of CS during 3 years. We also assessed relation between patients' background, serum concentration of TNF-α, IL-6, IL-10, IFX and clinical outcome.
Methods The study subjects were 21 patients with BD diagnosed based on the criteria recommended by the International Study Group for BD. The diagnosis of intestinal-BD was based on colonoscopy and it showed massive ulceration in the ileocecal region in all patients. The primary endpoint was the rate of disappearance of ileocecal ulceration at 1 year of therapy and secondary endpoints is the ameliorating effect based on the “Disease Activity Index for Intestinal Behçet's disease” (DAIBD)2), and the dose of CS tapered during 3-year treatment with IFX.
Results Baseline characteristics were as following; the mean age was 42.3, 5 male and 16 female, 5 patients were HLA-B51 positive. 10 patients had repeated relapses (range, 1-8 times), 6 patients experienced intestinal perforation, and 3 patients required surgical treatment before IFX therapy. We used 3-6mg/body of IFX with MTX (mean 9.72mg) to all of the patients. No severe adverse event was observed. Retention rate at 3 year treatment of IFX was 85.7% and 3 patients required switching to other TNF inhibitors due to inadequate response. The healing rate of ulceration evaluated by colonoscopy at 1 year was 76.2% (15 cases). DAIBD score and dose of CS were significantly decreased from 73.3 to 11.7, 14.2 to 1.3 mg/day respectively during 3y and 56.3% of the patients reached “Quiescent” defines as <19 of DAIBD at 1 year. DAIBD at baseline, the history of relapse associated with achieving Quiescent at 1 year, but sex, age, disease duration, HLA-B51, serum TNF, IL-6, IL-10 levels at the introduction of IFX did not. In this study, we experienced a patient who achieved clinical remission by IFX and it has been successfully maintained with only low dose of MTX for 5 years after discontinuation of IFX.
Conclusions IFX could ameliorate severe intestinal involvement in 15 out of 21 patients who are refractory to conventional therapy and suppress disease activity without relapse for 3 years despite of CS tapering. In addition, our case also suggests that the biologic-free remission is possible even in refractory intestinal-BD. However, this pilot study would warrant further investigation with more subjects and longer duration.
Iwata S et al. Rheumatology. 2009;48(8):1012-3.
Jae Hee Cheon et al. Inflamm Bowel Dis. 2011;17(2):605-13.
Disclosure of Interest None declared
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