Deficiency of glycosylphosphatidyl inositol-anchored proteins in patients with aplastic anaemia does not affect response to immunosuppressive therapy

Br J Haematol. 1998 Apr;101(1):90-3. doi: 10.1046/j.1365-2141.1998.00660.x.

Abstract

Deficient expression of glycosylphosphatidyl inositol (GPI)-anchored proteins in aplastic anaemia (AA) patients has previously been reported to be associated with a poor response to immunosuppressive (IS) therapy. Here we report the response to IS therapy of 111 patients with AA and correlate this with GPI-anchored protein expression on peripheral blood cells by flow cytometry. A GPI-anchored protein deficient population was identified in 15% (17/111) of patients with AA who had a negative Ham's test and no laboratory evidence of haemolysis. Patients were treated with antilymphocyte globulin and/or cyclosporin A, or oxymetholone. Bone marrow transplantation was performed in 12 patients, seven of whom had not responded to IS therapy. In patients tested for CPI-anchored protein expression prior to IS therapy there was no difference in response rate to IS therapy between AA patients with a GPI-anchored protein deficiency and those with normal GPI-anchored protein expression (50% response rate versus 75%, respectively). Survival in these two groups was similar at 90% with follow-up over 140 months from diagnosis. Eight of the 17 AA patients who developed a GPI-anchored protein-deficient population later went on to develop a positive Ham's test. From this study we demonstrate a lower incidence of GPI-anchored protein deficiency in AA patients compared with previous reports. In addition we have shown that the presence of a GPI-anchored protein-deficient cell population in patients with AA who have a negative Ham's test is not a poor prognostic factor in terms of response and survival after IS therapy.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Anemia, Aplastic / metabolism*
  • Anemia, Aplastic / therapy
  • Bone Marrow Transplantation
  • Child
  • Female
  • Glycosylphosphatidylinositols / deficiency*
  • Humans
  • Immunosuppression Therapy*
  • Male
  • Middle Aged
  • Prognosis
  • Survival Rate

Substances

  • Glycosylphosphatidylinositols