Glucocorticoid and cyclosporine refractory adult onset Still's disease successfully treated with tocilizumab

Clin Rheumatol. 2009 Apr;28(4):485-7. doi: 10.1007/s10067-009-1097-z. Epub 2009 Jan 30.

Abstract

We report a 29-year-old Japanese woman with disseminated intravascular coagulation (DIC) and adult onset Still's disease (AOSD). Her disease was refractory to high-dose glucocorticoids, two courses of steroid pulse therapy, and addition of cyclosporine (3.5 mg/kg/day). The serum interleukin-6 level was markedly elevated. Therefore, we administered an anti-interleukin-6 receptor antibody (tocilizumab, 8 mg/kg fortnightly), which dramatically improved her symptoms and the levels of acute-phase proteins. In addition, rapid tapering of the glucocorticoid dose was possible. Four months later, she was maintained on tocilizumab infusion once a month with low-dose steroid therapy. Cyclosporine is one of the first-line immunosuppressants for AOSD, especially when associated with DIC, hepatic failure, or hemophagocytic syndrome. In patients with cyclosporine-resistant AOSD, tocilizumab may be another useful option.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Antibodies, Monoclonal / pharmacology*
  • Antibodies, Monoclonal, Humanized
  • Cyclosporine / pharmacology*
  • Disseminated Intravascular Coagulation / diagnosis*
  • Disseminated Intravascular Coagulation / drug therapy
  • Female
  • Glucocorticoids / pharmacology*
  • Humans
  • Immunosuppressive Agents / pharmacology
  • Interleukin-6 / metabolism
  • Still's Disease, Adult-Onset / drug therapy*
  • Time Factors

Substances

  • Antibodies, Monoclonal
  • Antibodies, Monoclonal, Humanized
  • Glucocorticoids
  • Immunosuppressive Agents
  • Interleukin-6
  • Cyclosporine
  • tocilizumab