Allogeneic bone marrow transplantation for active Epstein-Barr virus-related lymphoproliferative disease and hemophagocytic lymphohistiocytosis in an infant with severe combined immunodeficiency syndrome

Bone Marrow Transplant. 2002 Mar;29(6):519-21. doi: 10.1038/sj.bmt.1703396.

Abstract

A 5-month-old male presented with fever, hepatosplenomegaly, leukocytosis with atypical lymphoblasts, anemia and thrombocytopenia. Severe combined imunodeficiency syndrome (T-, B+, NK+), B lymphoproliferative disease and hemophagocytic lymphohistiocytosis triggered by Epstein-Barr virus (EBV) were diagnosed. As his clinical situation deteriorated rapidly, BMT was performed with unmanipulated marrow stem cells from his EBV-positive HLA-identical sister after conditioning with dexamethasone (1.75 mg/kg/day), cyclophosphamide (114 mg/kg) and etoposide (10 mg/kg), with no immunosuppression given post transplant. Engraftment occurred on day 6 with explosive proliferation of donor CD8(+) T cells. The patient died 3 days later from acute respiratory distress syndrome. Autopsy revealed full donor engraftment and no signs of hemophagocytic lymphohistiocytosis or B lymphoproliferative disease. Thus, transplanted T cells can expand very rapidly within days after BMT and clear EBV lymphoproliferative disease and hemophagocytic lymphohistiocytosis.

Publication types

  • Case Reports

MeSH terms

  • Acute Disease
  • Bone Marrow Transplantation / methods*
  • Epstein-Barr Virus Infections / surgery*
  • Fatal Outcome
  • Herpesvirus 4, Human / isolation & purification*
  • Histiocytosis, Non-Langerhans-Cell / surgery*
  • Humans
  • Infant
  • Lymphoproliferative Disorders / diagnosis
  • Lymphoproliferative Disorders / surgery*
  • Lymphoproliferative Disorders / virology
  • Male
  • Severe Combined Immunodeficiency / surgery*
  • Severe Combined Immunodeficiency / virology
  • Transplantation, Homologous