Efficacy of cyclosporine A in the treatment of macrophage activation syndrome in juvenile arthritis: report of five cases

J Pediatr. 1996 Nov;129(5):750-4. doi: 10.1016/s0022-3476(96)70160-9.

Abstract

Objectives: To evaluate the efficacy of cyclosporine A in the treatment of macrophage activation syndrome (MAS) occurring in children with juvenile arthritis.

Study design: MAS developed in two boys and three girls with systemic juvenile arthritis (four) and polyarticular juvenile arthritis (one). In three children whose condition was life-threatening, increased parenteral administration of corticosteroids failed to improve their condition; therefore cyclosporine A (2 to 5 mg/kg per day) was added. In two other patients with less severe clinical manifestations, cyclosporine A alone (2 to 8 mg/kg per day) was given.

Results: After the introduction of cyclosporine A, rapid improvement was obtained in all patients and apyrexia occurred within 24 to 48 hours. The biologic abnormalities disappeared more slowly (up to 5 weeks for liver enzymes).

Conclusions: These observations underline the usefulness of cyclosporine A in this complication. The use of this drug may circumvent the need for increased doses of corticosteroids in some patients. The mechanism of action of cyclosporine A remains speculative, but these results indicate indirectly that T-helper lymphocytes may play a role in the pathogenesis of MAS.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Antirheumatic Agents / therapeutic use*
  • Arthritis, Juvenile / drug therapy*
  • Arthritis, Juvenile / physiopathology
  • Child
  • Child, Preschool
  • Cyclosporine / therapeutic use*
  • Female
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Macrophage Activation / drug effects*
  • Male
  • Syndrome
  • Treatment Outcome

Substances

  • Antirheumatic Agents
  • Immunosuppressive Agents
  • Cyclosporine