Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Institute of Human Genetics Munich, |
RCV001254094 | SCV001430014 | pathogenic | Nail-patella syndrome | 2020-07-16 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV001388093 | SCV001588942 | pathogenic | not provided | 2023-12-01 | criteria provided, single submitter | clinical testing | This sequence change replaces alanine, which is neutral and non-polar, with proline, which is neutral and non-polar, at codon 236 of the LMX1B protein (p.Ala236Pro). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individuals with clinical features of nail-patella syndrome (PMID: 9837817; Invitae). This variant is also known as p.Ala213Pro. ClinVar contains an entry for this variant (Variation ID: 976712). 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) performed at Invitae indicates that this missense variant is expected to disrupt LMX1B protein function with a positive predictive value of 80%. Experimental studies have shown that this missense change affects LMX1B function (PMID: 9837817). For these reasons, this variant has been classified as Pathogenic. |
Genetics and Molecular Pathology, |
RCV001254094 | SCV002557005 | pathogenic | Nail-patella syndrome | 2020-12-11 | criteria provided, single submitter | clinical testing | |
Gene |
RCV001388093 | SCV002599632 | pathogenic | not provided | 2023-06-23 | criteria provided, single submitter | clinical testing | Reported in unrelated individuals with nail-patella syndrome in the published literature (McIntosh et al., 1998); Published functional studies demonstrate A236P impairs LMX1B transactivation and acts through a haploinsufficiency mechanism (Dreyer et al., 2000); Not observed at significant frequency in large population cohorts (gnomAD); Also known as c.637 G>C p.A213P using alternate nomenclature; 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: 32963778, 10767331, 9837817) |
Fulgent Genetics, |
RCV002485994 | SCV002784339 | pathogenic | Nail-patella syndrome; Nail-patella-like renal disease | 2021-08-13 | criteria provided, single submitter | clinical testing |