Inherited disorders of the IL-12-IFN-gamma axis in patients with disseminated BCG infection.

Details

Serval ID
serval:BIB_E55A99E64269
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Inherited disorders of the IL-12-IFN-gamma axis in patients with disseminated BCG infection.
Journal
European journal of pediatrics
Author(s)
Mansouri D., Adimi P., Mirsaeidi M., Mansouri N., Khalilzadeh S., Masjedi M.R., Adimi P., Tabarsi P., Naderi M., Filipe-Santos O., Vogt G., de Beaucoudrey L., Bustamante J., Chapgier A., Feinberg J., Velayati A.A., Casanova J.L.
ISSN
0340-6199 (Print)
ISSN-L
0340-6199
Publication state
Published
Issued date
12/2005
Peer-reviewed
Oui
Volume
164
Number
12
Pages
753-757
Language
english
Notes
Publication types: Case Reports ; Journal Article
Publication Status: ppublish
Abstract
Disseminated BCG infection is a rare complication of vaccination that occurs in patients with impaired immunity. In recent years, a series of inherited disorders of the IL-12-IFN-gamma axis have been described that predispose affected individuals to disseminated disease caused by BCG, environmental Mycobacteria, and non-typhoidal Salmonella. The routine immunological work-up of these patients is normal and the diagnosis requires specific investigation of the IL-12-IFN-gamma circuit. We report here the first two such patients originating from and living in Iran. The first child is two years old and suffers from complete IFN-gamma receptor 2 deficiency and disseminated BCG infection. He is currently in clinical remission thanks to prolonged multiple antibiotic therapy. The other, a 28-year-old adult, suffers from IL-12p40 deficiency and presented with disseminated BCG infection followed by recurrent episodes of systemic salmonellosis. He is now doing well. A third patient of Iranian descent, living in North America, was reported elsewhere to suffer from IL-12Rbeta1 deficiency. These three patients thus indicate that various inherited defects of the IL-12-IFN-gamma circuit can be found in Iranian people. In conclusion we recommend to consider the disorders of the IL-12-IFN-gamma circuit in all patients with severe BCG infection, disseminated environmental mycobacterial disease, or systemic non-typhoidal salmonellosis, regardless of their ethnic origin and country of residence.
Keywords
Adult, BCG Vaccine/adverse effects, Child, Preschool, Genetic Predisposition to Disease, Humans, Interferon-gamma/genetics, Interleukin-12/genetics, Interleukin-12 Subunit p40, Iran, Male, Mycobacterium bovis, Protein Subunits/genetics, Receptors, Interferon/genetics, Salmonella Infections/genetics, Salmonella Infections/immunology, Tuberculosis/genetics, Tuberculosis/immunology, Tuberculosis/microbiology
Pubmed
Web of science
Create date
04/02/2021 20:04
Last modification date
07/07/2021 6:37
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