Variable outcome of experimental interferon-γ therapy of disseminated Bacillus Calmette-Guerin infection in two unrelated interleukin-12Rβ1-deficient Slovakian children View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2005-01-05

AUTHORS

Timo Ulrichs, Claire Fieschi, Eva Nevicka, Helmut Hahn, Martin Brezina, Stefan H. E. Kaufmann, Jean-Laurent Casanova, Klara Frecerova

ABSTRACT

Mycobacterium bovis Bacillus Calmette-Guerin (BCG) is an attenuated live vaccine that may cause life-threatening clinical disease in children with impaired immunity. In particular, patients with any of the nine known inherited disorders of the interleukin-12/23 interferon-γ (IL-12/23-IFNγ) axis are highly vulnerable to BCG. We describe two unrelated young Slovakian children suffering from disseminated BCG infection which developed shortly after routine BCG vaccination after birth. During treatment with selected anti-BCG antibiotics, resistance against several of these drugs developed. In both children, interleukin-12/23 receptor β1 (IL-12/23Rβ1) deficiency was diagnosed. Thus, in addition to chemotherapy, immunomodulatory treatment with recombinant IFN-γ was performed as the pathogenesis of BCG disease in IL-12Rβ1 deficiency involves impaired IL-12- and IL-23-dependent IFN-γ production by lymphocytes. One child responded to treatment and is presently doing well whereas the second patient died. Conclusion:The marked variability of outcome of disseminated Bacillus Calmette-Guerin disease in interleukin-12/23 receptor β1-deficient children sharing the same ethnic origin and exposed to a similar environment as presented in these case reports has to be taken into consideration for diagnosis and treatment of infections due to this genetic defect. More... »

PAGES

166-172

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00431-004-1599-2

DOI

http://dx.doi.org/10.1007/s00431-004-1599-2

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1031221986

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/15633050


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