Relapse of aplastic anemia responsive to sirolimus combined with cyclosporine

Pediatr Blood Cancer. 2011 Jul 1;56(7):1133-5. doi: 10.1002/pbc.22865. Epub 2011 Feb 11.

Abstract

Aplastic anemia (AA) is an immune-mediated disease. Although most patients are responsive to immunosuppressive therapy (IST) with a combination of anti-thymocyte globulin (ATG) and cyclosporine (CsA), some patients relapse or are refractory to IST. Sirolimus (rapamysin) inhibits the serine-threonine kinase mammalian target of rapamysin (mTOR), and blocks CsA-resistant and calcium-independent pathways late in the progression of the T-cell cycle. We report two cases of AA which relapsed after CsA and ATG plus CsA, respectively. They achieved transfusion independence after retreatment with sirolimus in combination with a CsA.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Anemia, Aplastic / drug therapy*
  • Antilymphocyte Serum / therapeutic use*
  • Child
  • Cyclosporine / therapeutic use*
  • Drug Therapy, Combination
  • Female
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Male
  • Recurrence
  • Sirolimus / therapeutic use*
  • Treatment Outcome

Substances

  • Antilymphocyte Serum
  • Immunosuppressive Agents
  • Cyclosporine
  • Sirolimus