Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Génétique des Maladies du Développement, |
RCV000760226 | SCV000890056 | pathogenic | Pitt-Hopkins syndrome; Corneal dystrophy, Fuchs endothelial, 3 | 2017-03-24 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV001855923 | SCV002278147 | likely pathogenic | Pitt-Hopkins syndrome | 2021-05-13 | criteria provided, single submitter | clinical testing | This variant is not present in population databases (ExAC no frequency). In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. Algorithms developed to predict the effect of missense changes on protein structure and function are either unavailable or do not agree on the potential impact of this missense change (SIFT: "Deleterious"; PolyPhen-2: "Probably Damaging"; Align-GVGD: "Class C0"). This variant has been observed in individual(s) with Pitt–Hopkins syndrome (PMID: 29695756). In at least one individual the variant was observed to be de novo. ClinVar contains an entry for this variant (Variation ID: 620020). This sequence change replaces arginine with tryptophan at codon 569 of the TCF4 protein (p.Arg569Trp). The arginine residue is highly conserved and there is a moderate physicochemical difference between arginine and tryptophan. |