Inherited disorders of the IL-12-INF-gamma axis in patients with disseminated BCG infection
D. Mansouri, P. Adimi, M. Mirsaeidi, P. Adimi, J. L. Casanova, M. R. Masjedi (Tehran, Islamic Republic Of Iran; Paris, France)
Source: Annual Congress 2006 - Pneumonia and other invasive pulmonary infections in children
Disease area: Respiratory infections
Abstract Disseminated BCG infection is rare complication of vaccination that accurs in patients with impaired immunity.In recent years a series of inherited disorders of the IL-12 INF-gamma axis have been described that predipose affected individuals to disseminated disease caused by BCG,environmental Mycobacteria,and non-typhoidal Salmonella. The routine immunological work-up of these patients are normal and the diagnosis requires specific investigation of the IL-12-INF-gamma circuit.We report here the first 3 such patients originating from and living in Iran.The first two child are 2 and 5 years old and suffer from complete INF-gamma receptor 2 deficiency and disseminated BCG infection.He and she are currently in clinicall remission thanks to prolonged antibiotic therapy.the Other,a 28 years old adult,suffer from IL-12p40 deficiency and presented with disseminated BCG infection followed by recurrent episodes of systemic salmonellosis.A fourth patientof Iranian decent,living in North America,was reported elsewhere to suffer from IL-12Rbetta1 deficiency.these patients thus indicates that varios inherited defects of IL-12-INF-gamma circuit can be found in Iranian people.In conclusion we recommended to consider the disorders of the IL12-INF-gamma circuit in all patients with severe BCG infection,disseminated environmental mycobacterial disease,or systemic non typhoidal salmonellosis,regardlless of their ethnic origion and country of residence.
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D. Mansouri, P. Adimi, M. Mirsaeidi, P. Adimi, J. L. Casanova, M. R. Masjedi (Tehran, Islamic Republic Of Iran; Paris, France). Inherited disorders of the IL-12-INF-gamma axis in patients with disseminated BCG infection. Eur Respir J 2006; 28: Suppl. 50, 1798
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