Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Baylor Genetics | RCV000445576 | SCV000485092 | pathogenic | Congenital heart disease; Failure to thrive; Abnormal skeletal morphology | 2016-12-11 | criteria provided, single submitter | clinical testing | This variant has been found four times in our laboratory in individuals with heart defects, skeletal anomalies, and failure to thrive. In two families, it co-segregated with disease, and it arose de novo in a third. |
Gene |
RCV001796027 | SCV002032419 | pathogenic | not provided | 2021-10-22 | criteria provided, single submitter | clinical testing | Published functional studies demonstrate that this variant leads to a gain of function with increased ABL kinase activity (Wang et al., 2017); Not observed in large population cohorts (Lek et al., 2016); This variant is associated with the following publications: (PMID: 28288113) |
MGZ Medical Genetics Center | RCV000496944 | SCV002581798 | likely pathogenic | Congenital heart defects and skeletal malformations syndrome | 2022-08-22 | criteria provided, single submitter | clinical testing | |
3billion | RCV000496944 | SCV005904148 | pathogenic | Congenital heart defects and skeletal malformations syndrome | 2023-10-05 | criteria provided, single submitter | clinical testing | The variant is not observed in the gnomAD v2.1.1 dataset. Predicted Consequence/Location: Missense variant Functional studies provide strong evidence of the variant having a damaging effect on the gene or gene product (PMID: 28288113, 28288113). In silico tool predictions suggest damaging effect of the variant on gene or gene product [REVEL: 0.67 (>=0.6, sensitivity 0.68 and specificity 0.92)]. Same nucleotide change resulting in same amino acid change has been previously reported as pathogenic/likely pathogenic with strong evidence (ClinVar ID: VCV000374795 /PMID: 28288113 /3billion dataset). The variant has been previously reported as de novo in a similarly affected individual (PMID: 28288113). Therefore, this variant is classified as Pathogenic according to the recommendation of ACMG/AMP guideline. |
OMIM | RCV000496944 | SCV000586799 | pathogenic | Congenital heart defects and skeletal malformations syndrome | 2017-08-02 | no assertion criteria provided | literature only |