Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
3billion | RCV005252289 | SCV005904358 | pathogenic | Severe intellectual disability-progressive spastic diplegia syndrome | 2023-10-13 | criteria provided, single submitter | clinical testing | The variant is not observed in the gnomAD v2.1.1 dataset. Predicted Consequence/Location: Missense changes are a common disease-causing mechanism. In silico tool predictions suggest damaging effect of the variant on gene or gene product [REVEL: 0.72 (>=0.6, sensitivity 0.68 and specificity 0.92); 3Cnet: 0.60 (>=0.6, sensitivity 0.72 and precision 0.9)]. Different nucleotide change resulting in same amino acid change has been previously reported as pathogenic/likely pathogenic with strong evidence (ClinVar ID: VCV000208674). The variant has been previously reported as de novo in a similarly affected individual (PMID: 33350591). Therefore, this variant is classified as Pathogenic according to the recommendation of ACMG/AMP guideline. |