Total submissions: 6
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Ce |
RCV001531491 | SCV001746658 | pathogenic | not provided | 2024-01-01 | criteria provided, single submitter | clinical testing | DCTN1: PP1:Strong, PM1, PM2, PM5, PS4:Moderate, PP3 |
Institute of Human Genetics Munich, |
RCV000020576 | SCV002764665 | pathogenic | Perry syndrome | 2020-09-25 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV003764613 | SCV004569188 | pathogenic | Amyotrophic lateral sclerosis type 1; Perry syndrome; Neuronopathy, distal hereditary motor, type 7B | 2023-06-15 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. 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 DCTN1 protein function. ClinVar contains an entry for this variant (Variation ID: 21390). This missense change has been observed in individuals with Perry syndrome (PMID: 19136952, 24343258, 28625595, 32717578). It has also been observed to segregate with disease in related individuals. This variant is not present in population databases (gnomAD no frequency). This sequence change replaces glycine, which is neutral and non-polar, with glutamic acid, which is acidic and polar, at codon 71 of the DCTN1 protein (p.Gly71Glu). |
Genomic Diagnostics Laboratory, |
RCV000020576 | SCV005039000 | pathogenic | Perry syndrome | 2024-05-02 | criteria provided, single submitter | clinical testing | Gly71Glu variant in DCTN1 has been reported previously in individuals with Perry síndrome (PMID: 19136952, 24343258, 28625595, 32717578). This variant is absent in population databases (gnomAD no frequency). This sequence change replaces glycine, which is neutral and non-polar, with glutamic acid, which is acidic and polar, at codon 71 of the DCTN1 protein (p.Gly71Glu). Acording to Farrer et al, 2009 (PMID: 19136952) functional analysis on a similar variant, Gly71Arg, revealed a modest decrease in microtubule binding comparable to Gly59Ser. We perfomed an in silico analyisis of this variant: Glu71 modifies the electrostatic potential of the CAP-Gly domain. Using ACMG Guidelines 2015, this is a pathogenic variant. |
OMIM | RCV000020576 | SCV000187718 | pathogenic | Perry syndrome | 2014-02-01 | no assertion criteria provided | literature only | |
Inherited Neuropathy Consortium Ii, |
RCV000020576 | SCV004174366 | uncertain significance | Perry syndrome | 2016-01-06 | no assertion criteria provided | literature only |