| Literature DB >> 23207905 |
Gabriella Vera1, Paola Rivera-Munoz, Vincent Abramowski, Laurent Malivert, Annick Lim, Christine Bole-Feysot, Christelle Martin, Benoit Florkin, Sylvain Latour, Patrick Revy, Jean-Pierre de Villartay.
Abstract
Cernunnos is a DNA repair factor of the nonhomologous end-joining machinery. Its deficiency in humans causes radiosensitive severe combined immune deficiency (SCID) with microcephaly, characterized in part by a profound lymphopenia. In contrast to the human condition, the immune system of Cernunnos knockout (KO) mice is not overwhelmingly affected. In particular, Cernunnos is dispensable during V(D)J recombination in lymphoid cells. Nevertheless, the viability of thymocytes is reduced in Cernunnos KO mice, owing to the chronic activation of a P53-dependent DNA damage response. This translates into a qualitative alteration of the T cell repertoire to one in which the most distal Vα and Jα segments are missing. This results in the contraction of discrete T cell populations, such as invariant natural killer T (iNKT) and mucosa-associated invariant T (MAIT) cells, in both humans and mice.Entities:
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Year: 2012 PMID: 23207905 PMCID: PMC3571340 DOI: 10.1128/MCB.01057-12
Source DB: PubMed Journal: Mol Cell Biol ISSN: 0270-7306 Impact factor: 4.272