Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000122469 | SCV002235250 | pathogenic | not provided | 2023-08-10 | criteria provided, single submitter | clinical testing | This sequence change replaces glycine, which is neutral and non-polar, with aspartic acid, which is acidic and polar, at codon 403 of the CACNA1D protein (p.Gly403Asp). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with CACNA1D-related conditions (PMID: 23913001, 28318089). In at least one individual the variant was observed to be de novo. ClinVar contains an entry for this variant (Variation ID: 66072). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) performed at Invitae indicates that this missense variant is expected to disrupt CACNA1D protein function. Experimental studies have shown that this missense change affects CACNA1D function (PMID: 23913001). For these reasons, this variant has been classified as Pathogenic. |
Athena Diagnostics | RCV000122469 | SCV002817245 | pathogenic | not provided | 2020-06-10 | criteria provided, single submitter | clinical testing | This variant occurs de novo in this current individual and in published literature (PMID: 28318089, 23913001). Assessment of experimental evidence suggests this is gain-of-function variant (PMID: 23913001).This variant has not been reported in large, multi-ethnic general populations (http://gnomad.broadinstitute.org).This observation is not an independent occurrence and has been identified in the same individual by RCIGM, the other laboratory participating in the GEMINI study. |
OMIM | RCV000056307 | SCV000087476 | pathogenic | Aldosterone-producing adenoma with seizures and neurological abnormalities | 2013-09-01 | no assertion criteria provided | literature only | |
Richard Lifton Laboratory, |
RCV000122469 | SCV000154977 | probable-pathogenic | not provided | no assertion criteria provided | not provided | Converted during submission to Likely pathogenic. | |
Division of Human Genetics, |
RCV000056307 | SCV000238465 | likely pathogenic | Aldosterone-producing adenoma with seizures and neurological abnormalities | 2015-01-14 | no assertion criteria provided | research | The variant (c.1208G>A) is likely pathogenic because it was previously reported as a de novo alteration in a patient with primary aldosteronism, seizures, and global developmental delays (Scholl et al. 2013, PMID: 23913001). Electrophysiological study of this variant showed that it was comparable to a different mutation at amino acid position 403 (p.Gly403Arg) found in an adrenal aldosterone-producing adenoma that is activated at less depolarizing potentials and impairs the inactivation of the wildtype allele (Scholl et al. 2013, PMID: 23913001). This variant does not occur in ExAC 0.3 even though this genomic region is well-covered. |