Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000430826 | SCV000517280 | likely pathogenic | not provided | 2023-10-23 | criteria provided, single submitter | clinical testing | Identified in individuals with Van der Woude spectrum in the published literature (PMID: 19282774, 29115498); Published functional studies demonstrate a damaging effect on periderm development (PMID: 28945736); Not observed at significant frequency in large population cohorts (gnomAD); This variant is associated with the following publications: (PMID: 19282774, 29115498, 28945736) |
Labcorp Genetics |
RCV002229128 | SCV000638964 | pathogenic | Orofacial cleft 6, susceptibility to; Popliteal pterygium syndrome; Van der Woude syndrome | 2021-09-26 | criteria provided, single submitter | clinical testing | Experimental studies have shown that this missense change affects IRF6 function (PMID: 28945736). This sequence change replaces glutamic acid with lysine at codon 404 of the IRF6 protein (p.Glu404Lys). The glutamic acid residue is highly conserved and there is a small physicochemical difference between glutamic acid and lysine. This variant is not present in population databases (ExAC no frequency). This missense change has been observed in individual(s) with Van der Woude syndrome (PMID: 19282774, 29115498). In at least one individual the variant was observed to be de novo. It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 217873). Algorithms developed to predict the effect of missense changes on protein structure and function (SIFT, PolyPhen-2, Align-GVGD) all suggest that this variant is likely to be disruptive. For these reasons, this variant has been classified as Pathogenic. |
Fulgent Genetics, |
RCV005016559 | SCV005645205 | likely pathogenic | Orofacial cleft 6, susceptibility to; Van der Woude syndrome 1; Autosomal dominant popliteal pterygium syndrome | 2024-01-04 | criteria provided, single submitter | clinical testing | |
Mendelics | RCV000201948 | SCV000256869 | pathogenic | Van der Woude syndrome 1 | 2014-06-02 | no assertion criteria provided | clinical testing |