Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000578629 | SCV000681131 | pathogenic | not provided | 2017-11-16 | criteria provided, single submitter | clinical testing | The Q203X variant in the PSAP gene has not been reported previously as a pathogenic variant nor as a benign variant, to our knowledge. This variant is predicted to cause loss of normal protein function either through protein truncation or nonsense-mediated mRNA decay. The Q203X variant is not observed in large population cohorts (Lek et al., 2016). We interpret Q203X as a pathogenic variant. |
Labcorp Genetics |
RCV002529049 | SCV003216605 | pathogenic | Sphingolipid activator protein 1 deficiency | 2023-04-19 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. ClinVar contains an entry for this variant (Variation ID: 489144). This variant has not been reported in the literature in individuals affected with PSAP-related conditions. This variant is not present in population databases (gnomAD no frequency). This sequence change creates a premature translational stop signal (p.Gln203*) in the PSAP gene. It is expected to result in an absent or disrupted protein product. Loss-of-function variants in PSAP are known to be pathogenic (PMID: 8554069, 11309366, 17616409, 19267410, 30632081). |
3billion | RCV002529049 | SCV003841900 | pathogenic | Sphingolipid activator protein 1 deficiency | 2023-02-23 | criteria provided, single submitter | clinical testing | The variant is not observed in the gnomAD v2.1.1 dataset. This variant was predicted to result in a loss or disruption of normal protein function through nonsense-mediated decay (NMD) or protein truncation. Multiple pathogenic variants are reported downstream of the variant. The variant has been reported to be associated with PSAP related disorder (ClinVar ID: VCV000489144). Therefore, this variant is classified as Pathogenic according to the recommendation of ACMG/AMP guideline. |