Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001881507 | SCV002151554 | uncertain significance | Holoprosencephaly sequence | 2021-03-26 | criteria provided, single submitter | clinical testing | This sequence change replaces methionine with threonine at codon 41 of the FOXH1 protein (p.Met41Thr). The methionine residue is highly conserved and there is a moderate physicochemical difference between methionine and threonine. While this variant is present in population databases (rs369306218), the frequency information is unreliable, as metrics indicate poor data quality at this position in the ExAC database. This variant has not been reported in the literature in individuals with FOXH1-related conditions. 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, but these predictions have not been confirmed by published functional studies and their clinical significance is uncertain. 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. |