Refractory disseminated coccidioidomycosis and mycobacteriosis in interferon-gamma receptor 1 deficiency

Clin Infect Dis. 2009 Sep 15;49(6):e62-5. doi: 10.1086/605532.

Abstract

Severe coccidioidomycosis is rare, and specific genetic susceptibility to the disease remains unidentified. We describe a patient with disseminated recalcitrant coccidioidomycosis with autosomal dominant interferon-gamma receptor 1 deficiency caused by a heterozygous IFNGR1 818del4 mutation. Therefore, the interleukin-12/interferon-gamma axis appears to be critical for control of coccidioidomycosis.

Publication types

  • Case Reports
  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, Non-P.H.S.

MeSH terms

  • Anti-Bacterial Agents / pharmacology
  • Anti-Bacterial Agents / therapeutic use
  • Antifungal Agents / pharmacology
  • Antifungal Agents / therapeutic use
  • Coccidioidomycosis / drug therapy*
  • Coccidioidomycosis / genetics
  • Coccidioidomycosis / immunology*
  • Drug Resistance, Bacterial
  • Drug Resistance, Fungal
  • Genes, Dominant
  • Genetic Predisposition to Disease / genetics*
  • Humans
  • Immunocompromised Host
  • Interferon gamma Receptor
  • Interferon-gamma / genetics
  • Interferon-gamma / metabolism
  • Interleukin-12 / genetics
  • Interleukin-12 / metabolism
  • Male
  • Mutation
  • Mycobacterium Infections / drug therapy*
  • Mycobacterium Infections / genetics
  • Mycobacterium Infections / immunology*
  • Receptors, Interferon / deficiency*
  • Receptors, Interferon / genetics*
  • Treatment Failure
  • Young Adult

Substances

  • Anti-Bacterial Agents
  • Antifungal Agents
  • Receptors, Interferon
  • Interleukin-12
  • Interferon-gamma