Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV002914783 | SCV003268300 | uncertain significance | not provided | 2023-07-03 | criteria provided, single submitter | clinical testing | This sequence change affects a donor splice site in intron 11 of the PRKCSH gene. It is expected to disrupt RNA splicing. Variants that disrupt the donor or acceptor splice site typically lead to a loss of protein function (PMID: 16199547), and loss-of-function variants in PRKCSH are known to be pathogenic (PMID: 12529853, 12577059, 20095989). In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. ClinVar contains an entry for this variant (Variation ID: 2057120). This variant has not been reported in the literature in individuals affected with PRKCSH-related conditions. This variant is present in population databases (rs373416744, gnomAD 0.1%), and has an allele count higher than expected for a pathogenic variant. |
Ambry Genetics | RCV004067120 | SCV005011678 | uncertain significance | Inborn genetic diseases | 2023-10-17 | criteria provided, single submitter | clinical testing | The c.1008+1G>A intronic variant results from a G to A substitution one nucleotide after coding exon 10 of the PRKCSH gene. Alterations that disrupt the canonical splice site are expected to cause aberrant splicing, resulting in an abnormal protein or a transcript that is subject to nonsense-mediated mRNA decay. Based on data from gnomAD, the A allele has an overall frequency of 0.013% (31/238808) total alleles studied. The highest observed frequency was 0.136% (23/16890) of East Asian alleles. This nucleotide position is highly conserved in available vertebrate species. In silico splice site analysis predicts that this alteration will weaken the native splice donor site. Based on insufficient or conflicting evidence, the clinical significance of this alteration remains unclear. |