Adjunctive treatment of disseminated Mycobacterium avium complex infection with interferon alpha-2b in a patient with complete interferon-gamma receptor R1 deficiency

Eur J Pediatr. 2007 Sep;166(9):981-5. doi: 10.1007/s00431-006-0339-1. Epub 2006 Nov 21.

Abstract

We report adjunct treatment of (interferon) IFN-alpha2b (Intron-A) in a patient with complete interferon-gamma receptor R1 (IFNGR1) deficiency suffering from disseminated infection with Mycobacterium avium complex (MAC) resistant to multiple anti-mycobacterial agents. A low dose of IFN-alpha2b (3 x 10(6) units/m(2) three times weekly subcutaneously) successfully attenuated progressive hepatosplenomegaly and abdominal/retroperitoneal/pelvic lymphadenopathy, although the patient continued to be mycobacteremic. This is the first report of a complete IFNGR1 deficiency treated with adjuvant IFN-alpha2b for disseminated MAC infection.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Drug Resistance, Multiple, Bacterial / immunology
  • Female
  • Humans
  • Immunologic Factors / therapeutic use*
  • Infant
  • Interferon alpha-2
  • Interferon-alpha / therapeutic use*
  • Mycobacterium avium Complex / immunology*
  • Mycobacterium avium Complex / pathogenicity
  • Mycobacterium avium-intracellulare Infection / drug therapy*
  • Mycobacterium avium-intracellulare Infection / immunology*
  • Receptors, Interferon / deficiency*
  • Recombinant Proteins
  • STAT1 Transcription Factor / immunology
  • Signal Transduction / immunology

Substances

  • Immunologic Factors
  • Interferon alpha-2
  • Interferon-alpha
  • Receptors, Interferon
  • Recombinant Proteins
  • STAT1 Transcription Factor
  • STAT1 protein, human
  • interferon gamma receptor