[Multifocal infection due to Mycobacterium intracellulare: first case of interferon gamma receptor partial dominant deficiency in tropical French territory]

Arch Pediatr. 2007 Mar;14(3):270-2. doi: 10.1016/j.arcped.2006.11.023. Epub 2007 Jan 12.
[Article in French]

Abstract

Nontuberculous mycobacterial infections are rare in immunocompetent children, and usually present as adenitis. We report a case of a 6-year-old girl with a multifocal chronic osteomyelitis and pulmonary localisation due to Mycobacterium intracellulare associated with an autosomal dominant mutation of interferon gamma receptor 1 gene (INFGR1) leading to a syndrome of mendelian predisposition to mycobacteria infections by partial deficiency of intracellular signalisation of gamma interferon. This child has been cured with anti-mycobacteria drugs and gamma interferon. This report focus on the importance of looking for a susceptibility of the host to infectious diseases, which can lead to a specific treatment. As far as we know, this is the first case described in a tropical area.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Female
  • France
  • Humans
  • Interferon gamma Receptor
  • Lung Diseases / microbiology
  • Mutation
  • Mycobacterium avium-intracellulare Infection / diagnosis*
  • Mycobacterium avium-intracellulare Infection / etiology
  • Osteomyelitis / complications
  • Osteomyelitis / microbiology
  • Receptors, Interferon / deficiency*
  • Receptors, Interferon / genetics
  • Respiratory Tract Infections / complications
  • Tropical Medicine

Substances

  • Receptors, Interferon