Etanercept for therapy-resistant macrophage activation syndrome

Pediatr Blood Cancer. 2008 Feb;50(2):419-21. doi: 10.1002/pbc.21019.

Abstract

Macrophage activation syndrome (MAS) is a severe, potentially fatal complication of childhood rheumatic diseases, especially systemic onset juvenile idiopathic arthritis (SoJIA). We report a 4-year-old girl with probable SoJIA who presented with MAS. She did not respond to pulse methyl prednisolone and Cyclosporine A (CsA). She also failed to respond to intravenous immunoglobulin (IVIG) therapy. Etanercept was started, based on the observation of increased serum levels of tumor necrosis factor-alpha (TNF-alpha) in patients with MAS. Her condition improved following etanercept, suggesting that etanercept might have a therapeutic role in resistant MAS.

MeSH terms

  • Child, Preschool
  • Etanercept
  • Female
  • Humans
  • Immunoglobulin G / therapeutic use*
  • Lymphohistiocytosis, Hemophagocytic / drug therapy*
  • Lymphohistiocytosis, Hemophagocytic / immunology
  • Macrophage Activation*
  • Receptors, Tumor Necrosis Factor / therapeutic use*

Substances

  • Immunoglobulin G
  • Receptors, Tumor Necrosis Factor
  • Etanercept