title: Fatal disseminated Mycobacterium smegmatis infection in a child with inherited interferon gamma receptor deficiency.
abstract: Mycobacterium smegmatis is a common environmental mycobacterium that was first identified in 1884, yet is a rare pathogen in humans. The few M. smegmatis infections reported to date have been localized and have occurred in association with a primary lesion in otherwise immunocompetent individuals. To our knowledge, no case of disseminated M. smegmatis infection has ever been reported, even in patients with severe immune deficiencies. We report a case of disseminated mycobacterial infection that was diagnosed in a 3-year-old girl. The pathogen was not identified as M. smegmatis until the patient was 6 years old. Her condition gradually worsened, and she died when she was 8 years old despite appropriate antimycobacterial therapy. No other opportunistic infections were documented. Immunological investigations revealed an inherited interferon gamma receptor 1 deficiency. This report identifies M. smegmatis as a new opportunistic agent that may be responsible for disseminated disease in immunocompromised individuals.
authors.affiliation: INSERM U429 and Service de Microbiologie, d'Anatomie Pathologique, and d'Immunologie Pédiatrique, Hôpital Necker-Enfants Malades, Paris, France.
authors.forename: C
authors.initials: C
authors.lastname: Pierre-Audigier
authors.forename: E
authors.initials: E
authors.lastname: Jouanguy
authors.forename: S
authors.initials: S
authors.lastname: Lamhamedi
authors.forename: F
authors.initials: F
authors.lastname: Altare
authors.forename: J
authors.initials: J
authors.lastname: Rauzier
authors.forename: V
authors.initials: V
authors.lastname: Vincent
authors.forename: D
authors.initials: D
authors.lastname: Canioni
authors.forename: J F
authors.initials: JF
authors.lastname: Emile
authors.forename: A
authors.initials: A
authors.lastname: Fischer
authors.forename: S
authors.initials: S
authors.lastname: Blanche
authors.forename: J L
authors.initials: JL
authors.lastname: Gaillard
authors.forename: J L
authors.initials: JL
authors.lastname: Casanova
mesh_terms: D016470:Bacteremia / Q000175:diagnosis / Q000276:immunology* / Q000382:microbiology; D002675:Child, Preschool; D017809:Fatal Outcome; D005260:Female; D006801:Humans; D016867:Immunocompromised Host; D009164:Mycobacterium Infections / Q000175:diagnosis / Q000276:immunology* / Q000382:microbiology; D009894:Opportunistic Infections / Q000175:diagnosis / Q000276:immunology* / Q000382:microbiology; D017471:Receptors, Interferon / Q000172:deficiency* / Q000235:genetics; D000096743:Interferon gamma Receptor
chemical_list: D017471:Receptors, Interferon; D000096743:Interferon gamma Receptor