Association of IRF5 polymorphisms with susceptibility to hemophagocytic lymphohistiocytosis in children

J Clin Immunol. 2011 Dec;31(6):946-51. doi: 10.1007/s10875-011-9583-x. Epub 2011 Sep 4.

Abstract

Introduction: Hemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory syndrome and has a varied genetic background. The polymorphism of interferon regulatory factor 5 gene (IRF5) was reported to be associated with susceptibility to macrophage activation syndrome. IRF5 acts as a master transcription factor in the activation of pro-inflammatory cytokines. We assessed associations of IRF5 gene polymorphisms with susceptibility to secondary HLH.

Methods: Three IRF5 single nucleotide polymorphisms (rs729302, rs2004640, and rs2280714) were genotyped using TaqMan assays in 82 secondary HLH patients and 188 control subjects.

Results: There was a significant association of the GT/TT genotype at rs2004640 with secondary HLH susceptibility (p < 0.01). The IRF5 haplotype (rs729302 A, rs2004640 T, and rs2280714 T) was associated with secondary HLH susceptibility (p < 0.01).

Conclusions: These findings indicate that IRF5 is a genetic factor influencing the susceptibility to secondary HLH and that the IRF5-associated immune response contributes to the pathogenesis of HLH.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Arthritis, Juvenile / genetics*
  • Arthritis, Juvenile / immunology
  • Arthritis, Juvenile / physiopathology
  • Child
  • Child, Preschool
  • Cytokines / genetics*
  • Cytokines / immunology
  • Cytokines / metabolism
  • DNA Mutational Analysis
  • Disease Progression
  • Female
  • Genetic Association Studies
  • Genetic Predisposition to Disease
  • Genotype
  • Humans
  • Infant
  • Inflammation Mediators / immunology
  • Inflammation Mediators / metabolism
  • Interferon Regulatory Factors / genetics*
  • Lymphohistiocytosis, Hemophagocytic
  • Macrophage Activation / genetics
  • Male
  • Polymorphism, Genetic
  • Transcriptional Activation / genetics

Substances

  • Cytokines
  • IRF5 protein, human
  • Inflammation Mediators
  • Interferon Regulatory Factors