Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000651145 | SCV000772995 | pathogenic | Severe combined immunodeficiency due to CARD11 deficiency; BENTA disease | 2022-01-06 | criteria provided, single submitter | clinical testing | Algorithms developed to predict the effect of missense changes on protein structure and function (SIFT, PolyPhen-2, Align-GVGD) all suggest that this variant is likely to be disruptive. For these reasons, this variant has been classified as Pathogenic. Experimental studies have shown that this missense change affects CARD11 function (PMID: 30170123). ClinVar contains an entry for this variant (Variation ID: 540972). This missense change has been observed in individual(s) with clinical features of autosomal dominant immunodeficiency with atopic disease (PMID: 30170123; Invitae). In at least one individual the variant was observed to be de novo. It has also been observed to segregate with disease in related individuals. This variant is present in population databases (rs765680532, gnomAD 0.01%). This sequence change replaces arginine, which is basic and polar, with histidine, which is basic and polar, at codon 47 of the CARD11 protein (p.Arg47His). |
Gene |
RCV004719916 | SCV005326075 | likely pathogenic | not provided | 2023-11-02 | criteria provided, single submitter | clinical testing | Published functional studies suggest a dominant negative effect (PMID: 30170123); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 33202260, 30170123) |