Epstein-Barr virus-induced hemophagocytic lymphohistiocytosis and X-linked lymphoproliferative disease: a mimicker of sepsis in the pediatric intensive care unit

Pediatrics. 2007 May;119(5):e1212-8. doi: 10.1542/peds.2006-1534. Epub 2007 Apr 2.

Abstract

A rare complication of infection with the Epstein-Barr virus is the development of hemophagocytic lymphohistiocytosis. Although most cases of Epstein-Barr virus-induced hemophagocytic lymphohistiocytosis develop in immunocompetent individuals, the rare immunodeficiency X-linked lymphoproliferative disease is often unmasked by Epstein-Barr virus infection and is clinically indistinguishable from Epstein-Barr virus-induced hemophagocytic lymphohistiocytosis. We describe the clinical course and management of a previously healthy 17-year-old boy who presented with hemodynamic collapse and severe systemic inflammatory response syndrome resulting from overwhelming hemophagocytosis in the setting of X-linked lymphoproliferative disease. A novel therapeutic approach using anti-tumor necrosis factor alpha therapy was instituted, aimed at attenuating the viral-induced hyperinflammatory state. Given the similarity to overwhelming sepsis, yet a substantially different therapeutic approach, this case illustrates the importance of early recognition and prompt treatment that are necessary to reduce the high morbidity and mortality associated with Epstein-Barr virus-induced hemophagocytic lymphohistiocytosis and X-linked lymphoproliferative disease.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Bone Marrow Transplantation
  • Diagnosis, Differential
  • Epstein-Barr Virus Infections / diagnosis*
  • Epstein-Barr Virus Infections / therapy
  • Etanercept
  • Herpesvirus 4, Human
  • Humans
  • Immunoglobulin G / therapeutic use
  • Intensive Care Units, Pediatric*
  • Lymphohistiocytosis, Hemophagocytic / diagnosis*
  • Lymphohistiocytosis, Hemophagocytic / therapy
  • Lymphoproliferative Disorders / diagnosis*
  • Lymphoproliferative Disorders / therapy
  • Male
  • Receptors, Tumor Necrosis Factor / therapeutic use
  • Sepsis / diagnosis*
  • Sepsis / therapy

Substances

  • Immunoglobulin G
  • Receptors, Tumor Necrosis Factor
  • Etanercept