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

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 o...

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Bibliographic Details
Published in:Archives de pédiatrie : organe officiel de la Société française de pédiatrie 2007-03, Vol.14 (3), p.270-272
Main Authors: Muszlak, M, Chapgier, A, Barry Harivelo, R, Castella, C, Crémades, F, Goulois, E, Laporte, R, Casanova, J-L, Ranaivoarivony, V, Hebert, J-C, Santiago, J, Picard, C
Format: Article
Language:fre
Subjects:
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
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Summary: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.
ISSN:0929-693X
1769-664X
DOI:10.1016/j.arcped.2006.11.023