Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV001658811 | SCV001873939 | likely pathogenic | not provided | 2024-04-18 | criteria provided, single submitter | clinical testing | In vivo and in vitro functional studies demonstrated that the presence of the P437L variant results in a dramatic decrease in enzymatic activity in comparison to wild-type (PMID: 27060699, 12807732); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 25952714, 27268095, 15723004, 19204079, 9497261, 16735994, 12807732, 15475877, 22128238, 19528825, 20660114, 25978063, 34528698, 35170016, 27060699, 27535533) |
Baylor Genetics | RCV003470876 | SCV004215486 | pathogenic | Anterior segment dysgenesis 6 | 2024-02-20 | criteria provided, single submitter | clinical testing | |
Revvity Omics, |
RCV001658811 | SCV004235476 | pathogenic | not provided | 2023-06-13 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV003594146 | SCV004292440 | pathogenic | Congenital glaucoma | 2024-03-10 | criteria provided, single submitter | clinical testing | This sequence change replaces proline, which is neutral and non-polar, with leucine, which is neutral and non-polar, at codon 437 of the CYP1B1 protein (p.Pro437Leu). This variant is present in population databases (rs56175199, gnomAD 0.01%). This missense change has been observed in individual(s) with nonsyndromic primary congenital glaucoma (PMID: 9497261, 12036985, 19234632). It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 1254629). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) has been performed at Invitae for this missense variant, however the output from this modeling did not meet the statistical confidence thresholds required to predict the impact of this variant on CYP1B1 protein function. Experimental studies have shown that this missense change affects CYP1B1 function (PMID: 27060699). For these reasons, this variant has been classified as Pathogenic. |
Fulgent Genetics, |
RCV005023212 | SCV005663679 | likely pathogenic | Glaucoma 3A; Anterior segment dysgenesis 6 | 2024-05-29 | criteria provided, single submitter | clinical testing |