Clinical and host genetic characteristics of Mendelian susceptibility to mycobacterial diseases in Japan

J Clin Immunol. 2011 Jun;31(3):309-14. doi: 10.1007/s10875-010-9498-y. Epub 2011 Jan 8.

Abstract

Purpose: The aim of this study is to investigate clinical characteristics and genetic backgrounds of Mendelian susceptibility to mycobacterial diseases (MSMD) in Japan.

Methods: Forty-six patients diagnosed as having MSMD were enrolled in this study. All patients were analyzed for the IFNGR1, IFNGR2, IL12B, IL12RB1, STAT1, and NEMO gene mutations known to be associated with MSMD.

Results: Six patients and one patient were diagnosed as having partial interferon-γ receptor 1 deficiency and nuclear factor-κB-essential modulator deficiency, respectively. Six of the seven patients had recurrent disseminated mycobacterial infections, while 93% of the patients without these mutations had only one episode of infection.

Conclusions: The patients with a genetic mutation were more susceptible to developing recurrent disseminated mycobacterial infections. Recurrent disseminated mycobacterial infections occurred in a small number of patients even without these mutations, suggesting the presence of as yet undetermined genetic factors underlying the development and progression of this disease.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Child, Preschool
  • Female
  • Flow Cytometry
  • Genetic Predisposition to Disease
  • Humans
  • I-kappa B Kinase / genetics*
  • Infant
  • Interferon gamma Receptor
  • Interferon-gamma / immunology
  • Japan / epidemiology
  • Longitudinal Studies
  • Male
  • Mendelian Randomization Analysis
  • Mutation*
  • Mycobacterium / physiology
  • Mycobacterium Infections / ethnology
  • Mycobacterium Infections / genetics*
  • Mycobacterium Infections / immunology
  • Mycobacterium Infections / microbiology
  • Mycobacterium Infections / pathology
  • Polymerase Chain Reaction
  • Receptors, Interferon / genetics*
  • Receptors, Interferon / immunology
  • Sequence Analysis, DNA

Substances

  • IKBKG protein, human
  • Receptors, Interferon
  • Interferon-gamma
  • I-kappa B Kinase