Background Adult Still’s disease (ASD) sometimes shows resistant to high-dose steroid and conventional immunosuppressive agents. Inflammatory cytokines seems to play a role in the pathogenesis1). Although several biologic agents have been used to treat refractory ASD2), their efficacy and safety are not established yet.
Objectives To examine the effectiveness and safely of several biologic agents, retrospective analysis was performed.
Methods We searched our data base for ASD patients who were diagnosed based on Yamaguchi’s criteria from August 2005 to January 2013. Clinical charts of the refractory ASD patients treated with any biologic agents were retrospectively reviewed. Clinical remission of ASD was defined if patients maintained all the following criteria, DAS28/CRP<2.3, no systemic symptoms and normal ferritin levels.
Results Refractory ASD was identified in 9 out of 84 ASD cases. Male/female ratio: 1:8, average age at 41.1±21.2 (20-76) years old, mean disease duration was 8.0±8.0 (3-29) years. They were treated with one or more biologics during the total 17 clinical courses. (Table) TNF antagonists (infliximab (IFX), adalimumab (ADA) and etanercept (ETN)) succeeded to induce clinical remission in 2 out of 10 clinical courses. Tocilizumab (TCZ) could induce clinical remission in 5 out of 7 clinical course. After switching from TNF antagonists to TCZ, 4 cases were effective out of 5. In the effective cases, the average remission maintenance period of anti-TNF was 28.5 months and TCZ was 40 months. During the treatment with TNF antagonists, severe adverse reaction was observed in 4 cases (ovarian cancer, severe hepatitis, cytomegalovirus infection and infusion reaction in each). No adverse effect was observed during TCZ therapy.
Conclusions TCZ seems to be highly effective and safe for the treatment of ASD. This hypothesis should be confirmed with larger clinical trials.
Hoshino T, Ohta A, Yang D, et al: Elevated serum interleukin 6, interferon-γ, and tumor necrosis factor-α levels in patients with adult Still’s disease. J Rheumatol 1998; 25: 396-8
De Bandt M, Saint-Marcoux B: Tocilizumab for multirefractory adult-onset Still’s disease. Ann Rheum Dis 2009; 68: 153-4
Disclosure of Interest None Declared
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