title: Immunosuppressive Therapy with Cyclosporin A of Severe Treatment Refractory Juvenile Rheumatoid Arthritis.
abstract: Rheumatoid arthritis is severe destructive inflammatory joint disease which usually leads to invalidisation of patient in spite of current therapies. The aim of the trial was to assess the effect of Cyclosporin A (Sandimmun (SIM), Sandos Pharma) in prevention of disability, reduction of inflammatory disease activity, tolerability, safety and suitability for a long treatment in severe treatment refractory juvenile rheumatoid arthritis (JRA). Fifteen patients of 7-15 years old (12 girls and 3 boys), of whom 6 had systemic seronegative and 9 had poliarticular seropositive severe JRA with anatomical stage 2-4 and functional capacities 2-3, were treated with Cyclosporin A in open self-controlled trial of 6 months duration for 15 patients and of 12 months duration for 7, All of them received non-steroid anti-inflammatory drugs and were regularly intra-articularly injected with corticosteroids and 6-prednisolone at a mean dose of 8.0 mg/day. Cyclosporin A was administrated in a dose of 3.5-4.5 mg/kg/day. Efficacy control was carried out according to dynamics of clinical and laboratory criteria of the disease activity, functional capacities and radiographic data. The results of the trial have shown that Cyclosporin A therapy led to statistically significant improvement in almost all clinical outcome variables (the number of swollen joints, p < 0.01, the number of painful joints, p < 0.001; pain score, p < 0.01; duration of morning stiffness, p < 0.001; volume of proximal interphalangeal joints, p < 0.001) and several laboratory parameters (ESR, p < 0.001; IgG level, p < 0.05; 6 seropositive patients became seronegative). All the patients exhibited significant increase in functional abilities and stabilization of joint destruction process. Response was considered as very good in 70% of patients and good in 20%, Prednisolone dose and the number of corticosteroid injections were reduced (p < 0.05). Adverse effects of Cyclosporin A included hypertrichosis, gingivitis, increase in appetite and weight, transitory increase in creatinin serum level of 30% over baseline, relapse of bacterial viral infection. These patients required temporal withdrawal of Cyclosporin A. It is concluded that Cyclosporin A therapy is effective in severe JRA, is well tolerated by patients, is suitable for a long-term treatment and has probable disease modifying effect in JRA.
authors.affiliation: Moscow Sechenov Medical Academy, Moscow, Russia.
authors.forename: Y. I.
authors.initials: YI
authors.lastname: Alexejeva
authors.forename: I. Y.
authors.initials: IY
authors.lastname: Shachbasjan
authors.forename: O. S.
authors.initials: OS
authors.lastname: Rosvadovscaja
authors.forename: V. Y.
authors.initials: VY
authors.lastname: Yeljashevich