Total submissions: 6
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
LDLR- |
RCV000237271 | SCV000296007 | pathogenic | Hypercholesterolemia, familial, 1 | 2016-03-25 | criteria provided, single submitter | literature only | |
Laboratory of Genetics and Molecular Cardiology, |
RCV000237271 | SCV000588666 | pathogenic | Hypercholesterolemia, familial, 1 | 2016-03-01 | criteria provided, single submitter | research | |
Labcorp Genetics |
RCV000816738 | SCV000957260 | pathogenic | Familial hypercholesterolemia | 2023-05-31 | criteria provided, single submitter | clinical testing | ClinVar contains an entry for this variant (Variation ID: 252336). Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may create or strengthen a splice site. This premature translational stop signal has been observed in individuals with hypercholesterolemia in a family and in several unrelated affected individuals (PMID: 7583548, 19013141, 21872251). It has also been observed to segregate with disease in related individuals. This variant is also known as K790X. For these reasons, this variant has been classified as Pathogenic. This sequence change creates a premature translational stop signal (p.Lys811*) in the LDLR gene. It is expected to result in an absent or disrupted protein product. Loss-of-function variants in LDLR are known to be pathogenic (PMID: 20809525, 28645073). This variant is not present in population databases (gnomAD no frequency). |
Ambry Genetics | RCV002450751 | SCV002737520 | pathogenic | Cardiovascular phenotype | 2020-09-30 | criteria provided, single submitter | clinical testing | The p.K811* pathogenic mutation (also known as c.2431A>T), located in coding exon 17 of the LDLR gene, results from an A to T substitution at nucleotide position 2431. This changes the amino acid from a lysine to a stop codon within coding exon 17. This alteration (also reported with legacy nomenclature as p.K790*) is one of the most common LDLR mutations found in Japan, and it has been detected in the heterozygous, compound heterozygous, and homozygous state in individuals with familial hypercholesterolemia (Maruyama T et al. Arterioscler. Thromb. Vasc. Biol., 1995 Oct;15:1713-8; Yu W et al. Atherosclerosis, 2002 Dec;165:335-42; Mabuchi H et al. Atherosclerosis, 2011 Feb;214:404-7). It has also been reported to segregate with disease in at least one family (Tada H et al. Atherosclerosis, 2011 Dec;219:663-6). Lymphocytes derived from patients heterozygous with this alteration exhibited approximately 55% of control LDLR activity (Tada H et al. Clin. Chim. Acta, 2009 Feb;400:42-7). In addition to the clinical data presented in the literature, 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. |
Women's Health and Genetics/Laboratory Corporation of America, |
RCV000816738 | SCV004240929 | pathogenic | Familial hypercholesterolemia | 2023-12-12 | criteria provided, single submitter | clinical testing | Variant summary: LDLR c.2431A>T (p.Lys811X) results in a premature termination codon, predicted to cause a truncation of the encoded protein or absence of the protein due to nonsense mediated decay, which are commonly known mechanisms for disease. The variant was absent in 251434 control chromosomes. c.2431A>T has been reported in the literature in multiple individuals affected with Familial Hypercholesterolemia and is one of the most common Japanese mutations (eg. Tada_2020, etc). Four submitters have cited clinical-significance assessments for this variant to ClinVar after 2014. All submitters classified the variant as pathogenic. Based on the evidence outlined above, the variant was classified as pathogenic. |
Laboratorium voor Moleculaire Diagnostiek Experimentele Vasculaire Geneeskunde, |
RCV000237271 | SCV000606652 | pathogenic | Hypercholesterolemia, familial, 1 | no assertion criteria provided | research |