Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Robarts Research Institute, |
RCV000408802 | SCV000484758 | likely pathogenic | Hypercholesterolemia, familial, 1 | criteria provided, single submitter | clinical testing | ||
Ambry Genetics | RCV002348123 | SCV002645967 | pathogenic | Cardiovascular phenotype | 2021-05-21 | criteria provided, single submitter | clinical testing | The p.E174* pathogenic mutation (also known as c.520G>T), located in coding exon 4 of the LDLR gene, results from a G to T substitution at nucleotide position 520. This changes the amino acid from a glutamic acid to a stop codon within coding exon 4. This alteration has been reported in a familial hypercholesterolemia (FH) cohort (Wang J et al. Arterioscler Thromb Vasc Biol, 2016 12;36:2439-2445). In addition, this alteration is expected to result in loss of function by premature protein truncation or nonsense-mediated mRNA decay. As such, this alteration is interpreted as a disease-causing mutation. |
Laboratory for Molecular Medicine, |
RCV004017597 | SCV004847687 | likely pathogenic | Homozygous familial hypercholesterolemia | 2019-04-16 | criteria provided, single submitter | clinical testing | The p.Glu174X variant in LDLR has been reported in the heterozygous state in 1 individual with coronary artery disease (Khera 2016) and 1 individual with familial hypercholesterolemia (FH; ClinVar submission accession: SCV000484758.1). It has also been identified in 0.007% (2/30616) of South Asian chromosomes by gnomAD (http://gnomad.broadinstitute.org). This nonsense variant leads to a premature termination codon at position 174, which is predicted to lead to a truncated or absent protein. Loss of function of the LDLR gene is an established disease mechanism in autosomal dominant FH. In summary, although additional studies are required to fully establish its clinical significance, this variant meets criteria to be classified as likely pathogenic for autosomal dominant FH. ACMG/AMP Criteria applied: PVS1, PM2. |