Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000310309 | SCV000933940 | likely pathogenic | Pitt-Hopkins-like syndrome 2 | 2024-07-19 | criteria provided, single submitter | clinical testing | This sequence change affects an acceptor splice site in intron 2 of the NRXN1 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 NRXN1 are known to be pathogenic (PMID: 19896112, 21964664, 23495017, 23533028, 25149956, 30031152). This variant is present in population databases (rs771759988, gnomAD 0.01%). Disruption of this splice site has been observed in individual(s) with clinical features of NRXN1-related conditions (PMID: 26185613, 33004838, 36368308). This variant is also known as g.51253608C>T. ClinVar contains an entry for this variant (Variation ID: 336552). Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. |
Gene |
RCV001753801 | SCV002006373 | uncertain significance | not provided | 2024-11-12 | criteria provided, single submitter | clinical testing | Identified as heterozygous in large cohorts of individuals with neurodevelopmental disorders and epilepsy, however detailed clinical and segregation information was not provided (Truty et al., 2019; Wang et al., 2020); Canonical splice site variant predicted to result in an in-frame loss of the adjacent exon in a gene for which loss of function is a known mechanism of disease; This variant is associated with the following publications: (PMID: 26185613, 31440721, 33004838, 36368308) |
Prevention |
RCV004530374 | SCV004115645 | likely pathogenic | NRXN1-related disorder | 2022-12-15 | criteria provided, single submitter | clinical testing | The NRXN1 c.773-1G>A variant is predicted to disrupt the AG splice acceptor site and interfere with normal splicing. This variant was reported as a rare loss of function variant in a study of individuals with autism spectrum disorder (Additional File 3, Griswold et al. 2015. PubMed ID: 26185613) and as a rare variant in individuals with neurodevelopmental disorders (Supplemental Dataset 5, Wang et al. 2020. PubMed ID: 33004838). This variant is reported in 0.011% of alleles in individuals of East Asian descent in gnomAD (http://gnomad.broadinstitute.org/variant/2-51253608-C-T). This variant is reported in 0.011% of alleles in individuals of East Asian descent in gnomAD (http://gnomad.broadinstitute.org/variant/2-51253608-C-T). Variants that disrupt the consensus splice acceptor site in NRXN1 are expected to be pathogenic. This variant is interpreted as likely pathogenic. |