Genes associated with common variable immunodeficiency: one diagnosis to rule them all?

DJA Bogaert, M Dullaers, BN Lambrecht… - Journal of medical …, 2016 - jmg.bmj.com
DJA Bogaert, M Dullaers, BN Lambrecht, KY Vermaelen, E De Baere, F Haerynck
Journal of medical genetics, 2016jmg.bmj.com
Common variable immunodeficiency (CVID) is a primary antibody deficiency characterised
by hypogammaglobulinaemia, impaired production of specific antibodies after immunisation
and increased susceptibility to infections. CVID shows a considerable phenotypical and
genetic heterogeneity. In contrast to many other primary immunodeficiencies, monogenic
forms count for only 2–10% of patients with CVID. Genes that have been implicated in
monogenic CVID include ICOS, TNFRSF13B (TACI), TNFRSF13C (BAFF-R), TNFSF12 …
Common variable immunodeficiency (CVID) is a primary antibody deficiency characterised by hypogammaglobulinaemia, impaired production of specific antibodies after immunisation and increased susceptibility to infections. CVID shows a considerable phenotypical and genetic heterogeneity. In contrast to many other primary immunodeficiencies, monogenic forms count for only 2–10% of patients with CVID. Genes that have been implicated in monogenic CVID include ICOS, TNFRSF13B (TACI), TNFRSF13C (BAFF-R), TNFSF12 (TWEAK), CD19, CD81, CR2 (CD21), MS4A1 (CD20), TNFRSF7 (CD27), IL21, IL21R, LRBA, CTLA4, PRKCD, PLCG2, NFKB1, NFKB2, PIK3CD, PIK3R1, VAV1, RAC2, BLK, IKZF1 (IKAROS) and IRF2BP2. With the increasing number of disease genes identified in CVID, it has become clear that CVID is an umbrella diagnosis and that many of these genetic defects cause distinct disease entities. Moreover, there is accumulating evidence that at least a subgroup of patients with CVID has a complex rather than a monogenic inheritance. This review aims to discuss current knowledge regarding the molecular genetic basis of CVID with an emphasis on the relationship with the clinical and immunological phenotype.
jmg.bmj.com