Severe disseminated mycobacterial infection in a boy with a novel mutation leading to IFN-γR2 deficiency

J Infect. 2012 Dec;65(6):568-72. doi: 10.1016/j.jinf.2012.08.008. Epub 2012 Aug 15.

Abstract

Mendelian susceptibility to mycobacterial diseases (MSMD) is a rare syndrome characterized by predisposition to severe, sometimes lethal, disease caused by otherwise poorly virulent mycobacteria. We report here a boy with a recurrent mycobacterial infection from the age of five months. Immunological analyses revealed an inability to respond to IFN-γ, subsequent genetic analyses revealed a novel homozygous mutation, r.679G > A in the IFNGR2 gene, resulting in a G227R substitution, that caused IFN-γR2 deficiency. This is only the 8th mutation in IFN-γR2 known so far. The boy eventually died of hepatic coma due to liver failure at the age of five.

Publication types

  • Case Reports

MeSH terms

  • Amino Acid Substitution
  • Child, Preschool
  • Fatal Outcome
  • Genetic Predisposition to Disease
  • Humans
  • Immunologic Deficiency Syndromes / genetics
  • Immunologic Deficiency Syndromes / immunology
  • Immunologic Deficiency Syndromes / metabolism
  • Infant
  • Interferon gamma Receptor
  • Male
  • Mutation
  • Mycobacterium Infections / genetics*
  • Mycobacterium Infections / immunology
  • Mycobacterium Infections / metabolism
  • Mycobacterium tuberculosis
  • Receptors, Interferon / deficiency*
  • Receptors, Interferon / genetics*
  • Receptors, Interferon / immunology

Substances

  • Receptors, Interferon