| Literature DB >> 25087226 |
Stuart E Turvey1, Anne Durandy2, Alain Fischer3, Shan-Yu Fung4, Raif S Geha5, Andreas Gewies6, Thomas Giese7, Johann Greil8, Bärbel Keller9, Margaret L McKinnon10, Bénédicte Neven11, Jacob Rozmus4, Jürgen Ruland12, Andrew L Snow13, Polina Stepensky14, Klaus Warnatz9.
Abstract
Next-generation DNA sequencing has accelerated the genetic characterization of many human primary immunodeficiency diseases (PIDs). These discoveries can be lifesaving for the affected patients and also provide a unique opportunity to study the effect of specific genes on human immune function. In the past 18 months, a number of independent groups have begun to define novel PIDs caused by defects in the caspase recruitment domain family, member 11 (CARD11)-B-cell chronic lymphocytic leukemia/lymphoma 10 (BCL10)-mucosa-associated lymphoid tissue lymphoma translocation gene 1 (MALT1 [CBM]) signalosome complex. The CBM complex forms an essential molecular link between the triggering of cell-surface antigen receptors and nuclear factor κB activation. Germline mutations affecting the CBM complex are now recognized as the cause of novel combined immunodeficiency phenotypes, which all share abnormal nuclear factor κB activation and dysregulated B-cell development as defining features. For this "Current perspectives" article, we have engaged experts in both basic biology and clinical immunology to capture the worldwide experience in recognizing and managing patients with PIDs caused by CBM complex mutations.Entities:
Keywords: CARD11-BCL10-MALT1 signalosome complex; CARMA1; combined immunodeficiency; congenital B-cell lymphocytosis; next-generation sequencing; nuclear factor κB; paracaspase; primary immunodeficiency diseases
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Year: 2014 PMID: 25087226 PMCID: PMC4167767 DOI: 10.1016/j.jaci.2014.06.015
Source DB: PubMed Journal: J Allergy Clin Immunol ISSN: 0091-6749 Impact factor: 10.793