Epstein-Barr virus-associated haemophagocytic lymphohistiocytosis in Wiskott-Aldrich syndrome

Acta Paediatr. 2003 Jul;92(7):859-61. doi: 10.1080/08035250310003631.

Abstract

A patient with Wiskott-Aldrich syndrome who developed Epstein-Barr virus-associated haemophagocytic lymphohistiocytosis (EBV-HLH) is described in this study. At 4 mo of age the patient developed fever associated with bicytopenia and splenomegaly. Analysis of a bone marrow specimen revealed extensive haemophagocytosis, and in situ hybridization for EBV of the bone marrow specimen using an EBV-encoded RNA probe was positive. Diagnosis of EBV-HLH was established and immunotherapy with HLH-94 protocol was started. HLH has been described in patients with other well-defined primary immunodeficiencies such as X-linked lymphoproliferative syndrome, Chediak-Higashi syndrome and Griscelli disease. Also, HLH was reported recently in severe combined immunodeficiency and DiGeorge syndrome.

Conclusion: The possibility of an underlying primary immunodeficiency should be considered in paediatric patients who present with HLH during infancy.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Epstein-Barr Virus Infections / drug therapy
  • Epstein-Barr Virus Infections / immunology
  • Epstein-Barr Virus Infections / virology*
  • Hepatomegaly / complications
  • Histiocytosis, Non-Langerhans-Cell / complications*
  • Histiocytosis, Non-Langerhans-Cell / virology*
  • Humans
  • Immunoglobulin A / immunology
  • Immunoglobulin G / immunology
  • Immunoglobulin M / immunology
  • Immunoglobulins, Intravenous / therapeutic use
  • Male
  • Splenomegaly / complications
  • Wiskott-Aldrich Syndrome / complications*

Substances

  • Immunoglobulin A
  • Immunoglobulin G
  • Immunoglobulin M
  • Immunoglobulins, Intravenous