Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000430933 | SCV000517011 | pathogenic | not provided | 2015-05-26 | criteria provided, single submitter | clinical testing | The T195I variant in the ACTG2 gene has not been reported previously as a pathogenic variant nor as a benign polymorphism, to our knowledge. The T195I substitution was not observed in approximately6500 individuals of European and African American ancestry in the NHLBI Exome Sequencing Project,indicating it is not a common benign variant in these populations. The T195I variant is a non-conservative amino acid substitution, which is likely to impact secondary protein structure as these residues differ inpolarity, charge, size and/or other properties. This substitution occurs at a position that is conserved acrossspecies. In silico analysis predicts this variant is probably damaging to the protein structure/function.Missense variants in nearby residues (G198D) have been reported in the Human Gene MutationDatabase in association with megacystic-microcolon-intestinal hypoperistalsis syndrome (Stenson et al., 2014), supporting the functional importance of this region of the protein. We interpret T195I as a pathogenic variant. |
Prevention |
RCV004529572 | SCV004109932 | uncertain significance | ACTG2-related disorder | 2022-12-30 | criteria provided, single submitter | clinical testing | The ACTG2 c.584C>T variant is predicted to result in the amino acid substitution p.Thr195Ile. This variant was reported in the heterozygous state in an individual with chronic intestinal pseudo-obstruction (CIPO, Patient 6, Moreno et al. 2016. PubMed ID: 27481187). This variant has not been reported in a large population database (http://gnomad.broadinstitute.org), indicating this variant is rare and is classified as pathogenic by one outside laboratory in ClinVar (https://www.ncbi.nlm.nih.gov/clinvar/variation/379697/). Although we suspect that this variant may be pathogenic, at this time, the clinical significance of this variant is uncertain due to the absence of conclusive functional and genetic evidence. |
OMIM | RCV001535811 | SCV001752410 | pathogenic | Visceral myopathy 1 | 2021-07-14 | no assertion criteria provided | literature only |