Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
LDLR- |
RCV000003943 | SCV000294388 | likely pathogenic | Hypercholesterolemia, familial, 1 | 2016-03-25 | criteria provided, single submitter | literature only | |
Cardiovascular Research Group, |
RCV000003943 | SCV000599300 | likely pathogenic | Hypercholesterolemia, familial, 1 | 2016-03-01 | criteria provided, single submitter | curation | |
Labcorp Genetics |
RCV002512730 | SCV003472875 | uncertain significance | Familial hypercholesterolemia | 2023-02-22 | criteria provided, single submitter | clinical testing | 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. Studies have shown that this variant alters LDLR gene expression (PMID: 14616764, 17625505). ClinVar contains an entry for this variant (Variation ID: 3745). This variant is also known as c.-45del. This variant has been observed in individual(s) with familial hypercholesterolemia (PMID: 14616764). It has also been observed to segregate with disease in related individuals. This variant is not present in population databases (gnomAD no frequency). This variant occurs in a non-coding region of the LDLR gene. It does not change the encoded amino acid sequence of the LDLR protein. |
All of Us Research Program, |
RCV000003943 | SCV004840284 | likely pathogenic | Hypercholesterolemia, familial, 1 | 2023-12-18 | criteria provided, single submitter | clinical testing | This variant deletes one nucleotide at the -138 position in the conserved SP1 binding site in the promoter region of the LDLR gene. This variant is also known as c.-45del and FH-Pyrgos in the literature. Functional studies using lymphocytes derived from a heterozygous individual have shown that this variant resulted in reduced LDLR activity and absent mRNA transcription (PMID: 14616764). An in-vitro functional study using transfected mammalian cells has shown that this variant causes a significant reduction in promoter activity (PMID: 14616764, 17625505). This variant has been reported in at least two unrelated individuals affected with familial hypercholesterolemia (PMID: 14616764, 35631530). It has been shown that this variant segregates with disease in three affected individuals in one family (PMID: 14616764). This variant has not been identified in the general population by the Genome Aggregation Database (gnomAD). Based on the available evidence, this variant is classified as Likely Pathogenic. |
OMIM | RCV000003943 | SCV000024108 | pathogenic | Hypercholesterolemia, familial, 1 | 2003-11-01 | no assertion criteria provided | literature only |