Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Revvity Omics, |
RCV000008930 | SCV002023916 | likely pathogenic | Dubin-Johnson syndrome | 2020-07-22 | criteria provided, single submitter | clinical testing | |
Prevention |
RCV003421913 | SCV004118106 | pathogenic | ABCC2-related disorder | 2023-07-30 | criteria provided, single submitter | clinical testing | The ABCC2 c.3449G>A variant is predicted to result in the amino acid substitution p.Arg1150His. This variant has been reported in the compound heterozygous state in two sibling with Dubin-Johnson syndrome, and also in the homozygous state in 5 individuals from 4 families with Dubin-Johnson syndrome (Mor-Cohen et al. 2001. PubMed ID: 11477083). In vitro functional studies suggest that this variant impairs the transport activity of ABCC2 (Mor-Cohen et al. 2001. PubMed ID: 11477083). This variant is interpreted as pathogenic. |
Labcorp Genetics |
RCV003574699 | SCV004354556 | pathogenic | not provided | 2024-02-18 | criteria provided, single submitter | clinical testing | This sequence change replaces arginine, which is basic and polar, with histidine, which is basic and polar, at codon 1150 of the ABCC2 protein (p.Arg1150His). This variant is present in population databases (rs72558200, gnomAD 0.01%). This missense change has been observed in individual(s) with Dubin-Johnson syndrome (PMID: 11477083). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 8419). 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 not expected to disrupt ABCC2 protein function with a negative predictive value of 80%. Experimental studies have shown that this missense change affects ABCC2 function (PMID: 11477083). For these reasons, this variant has been classified as Pathogenic. |
Fulgent Genetics, |
RCV000008930 | SCV005664894 | likely pathogenic | Dubin-Johnson syndrome | 2024-05-01 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000008930 | SCV000029140 | pathogenic | Dubin-Johnson syndrome | 2001-10-05 | no assertion criteria provided | literature only |