Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
3billion | RCV000003662 | SCV002058235 | pathogenic | Nephrotic syndrome, type 4 | 2022-01-03 | criteria provided, single submitter | clinical testing | The variant has been previously reported as de novo in a similarly affected individual (PMID: 9529364, PS2_S). Same nucleotide change resulting in same amino acid change has been previously reported to be associated with WT1 related disorder (ClinVar ID: VCV000003490, PMID:1655284). A different missense change at the same codon (p.Asp469Gly, p.Asp469Tyr) has been reported as pathogenic/likely pathogenic with strong evidence (ClinVar ID: VCV000003489,VCV000547167, PMID:22876585, PMID:1655284, PMID:10738002, PMID:24402088, PM5_M). In silico tool predictions suggest damaging effect of the variant on gene or gene product (3CNET: 0.895, PP3_P). A missense variant is a common mechanism associated with Nephrotic syndrome (PP2_P). It is observed at an extremely low frequency in the gnomAD v2.1.1 dataset (total allele frequency: 0.000000, PM2_M). Therefore, this variant is classified as pathogenic according to the recommendation of ACMG/AMP guideline. |
Gene |
RCV003322746 | SCV004028420 | pathogenic | not provided | 2023-08-15 | criteria provided, single submitter | clinical testing | Published functional studies demonstrate a damaging effect: decreased DNA binding affinity (Pelletier et al, 1991); Not observed at significant frequency in large population cohorts (gnomAD); Also known as c.754G>A p.D252N, c.1186G>A p.D396N, and c.1405G>A p.D469N; This variant is associated with the following publications: (PMID: 24379226, 31816618, 1338906, 1655284, 25818337, 29869118, 29294058, 9529364, 9090524, 32891756, 32053599, 34031707, 31328266, 33392118, 25077094, 20442690, Bekheirnia2021[article], 15665984) |
OMIM | RCV000003661 | SCV000023824 | pathogenic | Drash syndrome | 1998-04-01 | no assertion criteria provided | literature only | |
OMIM | RCV000003662 | SCV000023825 | pathogenic | Nephrotic syndrome, type 4 | 1998-04-01 | no assertion criteria provided | literature only |