Thalidomide therapy for a girl with systemic-onset　juvenile　idiopathic arthritis　showing　refractory　arthritis　and hypercytokinemia．

Abstract

We report a case of a five-year-old girl suffering from systemic-onset juvenile idiopathic arthritis (S-JIA). Her joint symptoms and hypercytokinemia persisted despite treatment with tocilizumab plus cyclosporine A or tacrolimus. In order to decrease the steroid dose, we introduced thalidomide in her treatment. Some foreign reports have indicated the effectiveness of 23 mg/kg of thalidomide, but in our case, even 5 mg /kg of thalidomide was not sufficiently effective to decrease the steroid dose, though serum IL-18 did decrease slightly. Subtle constipation was noticed during thalidomide treatment, but no other side effects, including peripheral neuropathy, were evident. When the efficacy of tocilizumab for S-JIA is not enough, we must grope for another means of treatment, and it appears that thalidomide, with meticulous attention to side effects, can be an option, although it is not clear why the drug was not significantly effective in our case.