Autologous stem cell transplantation for refractory juvenile idiopathic arthritis: analysis of clinical effects, mortality, and transplant related morbidity

Ann Rheum Dis. 2004 Oct;63(10):1318-26. doi: 10.1136/ard.2003.017798.

Abstract

Objective: To evaluate the safety and efficacy of autologous stem cell transplantation (ASCT) for refractory juvenile idiopathic arthritis (JIA).

Design: Retrospective analysis of follow up data on 34 children with JIA who were treated with ASCT in nine different European transplant centres. Rheumatological evaluation employed a modified set of core criteria. Immunological reconstitution and infectious complications were monitored at three month intervals after transplantation.

Results: Clinical follow up ranged from 12 to 60 months. Eighteen of the 34 patients (53%) with a follow up of 12 to 60 months achieved complete drug-free remission. Seven of these patients had previously failed treatment with anti-TNF. Six of the 34 patients (18%) showed a partial response (ranging from 30% to 70% improvement) and seven (21%) were resistant to ASCT. Infectious complications were common. There were three cases of transplant related mortality (9%) and two of disease related mortality (6%).

Conclusions: ASCT in severely ill patients with JIA induces a drug-free remission of the disease and a profound increase in general wellbeing in a substantial proportion of patients, but the procedure carries a significant mortality risk. The following adjustments are proposed for future protocols: (1) elimination of total body irradiation from the conditioning regimen; (2) prophylactic administration of antiviral drugs and intravenous immunoglobulins until there is a normal CD4+ T cell count.

Publication types

  • Evaluation Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Arthritis, Juvenile / immunology
  • Arthritis, Juvenile / therapy*
  • Child
  • Child, Preschool
  • Female
  • Follow-Up Studies
  • Graft Survival
  • Health Status Indicators
  • Humans
  • Infant
  • Male
  • Opportunistic Infections / etiology
  • Patient Selection
  • Retrospective Studies
  • Severity of Illness Index
  • Stem Cell Transplantation / adverse effects
  • Stem Cell Transplantation / methods*
  • Survival Analysis
  • T-Lymphocyte Subsets / immunology
  • Transplantation Conditioning / methods
  • Transplantation, Autologous
  • Treatment Outcome