Abstract
Purpose:
We have previously reported the short-term results of anti-TNF therapy in the pediatric population with severe uveitis. Our aim is to evaluate the mid-term efficacy and safety of adalimumab in the same population.
Methods:
We retrospectively analysed 15 children with severe uveitis, who were treated with adalimumab (20 or 40-mg, every two weeks) when the previous immunosuppressive therapy has been ineffective. It consisted of systemic corticosteroids (n=15), methotrexate (n=15), azathioprine (n=5), mycophenolate mofetil (n=1), infliximab (n=6), etanercept (n=6), interferon-alpla (n=1). The etiology of uveitis was juvenile idiopathic arthritis (n = 11), sarcoidosis (n = 1), spondyloarthropathy (n = 1), idiopathic or unknown (n = 2). Primary outcome was to assess laser flare photometry values after adalimumab therapy compared with baseline. Clinical features (Standardization of Uveitis Nomenclature criteria, macular edema and papillitis), oral prednisone threshold, visual acuity, side effects and complications of treatment were also considered.
Results:
Median age was 13.1 years (range 6-20.8) and sex ratio (F/M) was 3. Median duration before adalimumab therapy was 82.6 months (range 16-262). The mean follow-up was 35.5 months (range 4-63) and the final median laser flare photometry value was significantly reduced from 149.5 ph/ms (range 24-335) to 85.4 ph/ms (range 4-224) p<0.005. Median oral prednisone decreased from 10.3 mg/day (range 0-30) to 3.2 mg/day (range 0-15) p<0.05. Uveitis was controlled in 11 cases (73.3%). Relapses occurred in one case (6.7%), adalimumab was ineffective in one case (6.7%) and was stopped in a patient who had excellent control of inflammation. Three children (20%) discontinued treatment due to severe side effects : one due to intolerance with an allergic reaction, one due to neurologic side-effects (optic neuropathy, polyneuropathy), one because of behavioral disorders.
Conclusions:
Adalimumab appears to be an effective and well tolerated treatment for refractory pediatric uveitis, with prolonged control of inflammation over several years, even after failure of other anti-TNF alpha agents. A prospective randomized double blind study is currently ongoing in order to confirm these retrospective results.
Keywords: 746 uveitis-clinical/animal model •
466 clinical (human) or epidemiologic studies: treatment/prevention assessment/controlled clinical trials •
555 immunomodulation/immunoregulation