Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000657979 | SCV000779750 | likely pathogenic | not provided | 2018-05-29 | criteria provided, single submitter | clinical testing | The c.6992+5G>A variant in the LAMA2 gene has been reported previously in the heterozygous state with a second LAMA2 variant in an individual with limb girdle muscular dystrophy, however, segregation studies to confirm phase were only performed on one parent (Harris et al., 2017). This variant is predicted to destroy the splice donor site in intron 49. Both in silico predictors and evolutionary conservation support a deleterious effect in this gene for which loss-of-function is a known mechanism for disease. The c.6992+5G>A variant is not observed at a significant frequency in large population cohorts (Lek et al., 2016). We interpret c.6992+5G>A as a likely pathogenic variant. |
Counsyl | RCV000673048 | SCV000798214 | uncertain significance | Merosin deficient congenital muscular dystrophy | 2018-03-01 | criteria provided, single submitter | clinical testing | |
Prevention |
RCV004527719 | SCV004108465 | uncertain significance | LAMA2-related disorder | 2023-03-20 | criteria provided, single submitter | clinical testing | The LAMA2 c.6992+5G>A variant is predicted to interfere with splicing. This variant has been reported in the heterozygous state (along with a second variant) in an individual with white matter changes (Table 1, individual 32, Harris et al. 2017. PubMed ID: 28877744). A skin biopsy for this individual demonstrated partial absence of laminin a2. In the gnomAD public population database this variant has been reported in one of ~250,000 alleles (https://gnomad.broadinstitute.org/variant/6-129781474-G-A), and is interpreted as uncertain significance and likely pathogenic in ClinVar (https://www.ncbi.nlm.nih.gov/clinvar/variation/546142/). At this time, the clinical significance of this variant is uncertain due to the absence of conclusive functional and genetic evidence. |
Labcorp Genetics |
RCV005091890 | SCV005834520 | uncertain significance | LAMA2-related muscular dystrophy | 2024-04-04 | criteria provided, single submitter | clinical testing | This sequence change falls in intron 49 of the LAMA2 gene. It does not directly change the encoded amino acid sequence of the LAMA2 protein. It affects a nucleotide within the consensus splice site. This variant is present in population databases (no rsID available, gnomAD 0.003%). This variant has been observed in individual(s) with limb girdle muscular dystrophy (PMID: 28877744). ClinVar contains an entry for this variant (Variation ID: 546142). Variants that disrupt the consensus splice site are a relatively common cause of aberrant splicing (PMID: 17576681, 9536098). 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 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. |
Institute of Human Genetics, |
RCV004813130 | SCV005438221 | likely pathogenic | Muscular dystrophy, limb-girdle, autosomal recessive 23 | no assertion criteria provided | clinical testing |