Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Revvity Omics, |
RCV003138653 | SCV003820067 | uncertain significance | Dopa-responsive dystonia due to sepiapterin reductase deficiency | 2022-08-09 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV003778793 | SCV004612149 | pathogenic | Dystonic disorder | 2023-09-22 | criteria provided, single submitter | clinical testing | ClinVar contains an entry for this variant (Variation ID: 2436316). For these reasons, this variant has been classified as Pathogenic. This variant disrupts a region of the SPR protein in which other variant(s) (p.Lys251*) have been determined to be pathogenic (PMID: 16917893, 18502672, 21431957, 21677200, 24212389, 25763508, 29116116). This suggests that this is a clinically significant region of the protein, and that variants that disrupt it are likely to be disease-causing. This variant has not been reported in the literature in individuals affected with SPR-related conditions. This variant is not present in population databases (gnomAD no frequency). This sequence change creates a premature translational stop signal (p.Gln206*) in the SPR gene. While this is not anticipated to result in nonsense mediated decay, it is expected to disrupt the last 56 amino acid(s) of the SPR protein. |