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Rheumatology 1999; 38: 764-766
© 1999 British Society for Rheumatology

Paediatric Rheumatology: Autologous Stem Cell Transplantation in Rheumatic Diseases of Childhood

Autologous stem cell transplantation: a possible treatment for refractory juvenile chronic arthritis?

Series Editor: P. Woo

N. M. Wulffraat and W. Kuis

Department of Paediatric Immunology, University Hospital for Children, `Het Wilhelmina Kinderziekenhuis', PO Box 85090, 3508 AB Utrecht, The Netherlands

Correspondence to: N. Wulffraat.

Abstract

Objective. In adults, autologous stem cell transplantation (ASCT) has been described recently as a possible treatment for severe autoimmune disease refractory to conventional treatment. We report here the four first children with severe forms of juvenile chronic arthritis (JCA) treated with ASCT.

Methods. We studied three children with systemic JCA and one child with polyarticular JCA. Unprimed bone marrow was harvested 1 month prior to ASCT. T-cell depletion of the graft was performed with CD2 and CD3 antibodies. We used a preparative regimen of antithymocyte globulin (ATG; 20 mg/kg), cyclophosphamide (Cy; 200 mg/kg) and low-dose total body irradiation (TBI; 4 Gy). Methotrexate (MTX) and cyclosporin A (CsA) were stopped before ASCT; prednisone was tapered after 2 months.

Results. After ASCT, our patients showed an anti-inflammatory-drug-free follow-up of 6–18 months with a marked decrease in joint swelling, pain and morning stiffness. The erythrocyte sedimentation rate (ESR), C-reactive protein (CRP) and haemoglobin (Hb) returned to near-normal values within 6 weeks. Despite T-cell depletion, there was a very rapid immune reconstitution. Two patients developed a limited varicella zoster virus (VZV) eruption which was treated by acyclovir.

KEY WORDS: Juvenile chronic arthritis, Refractory, Treatment, Autologous stem cell transplantation


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