Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000493351 | SCV000583363 | uncertain significance | not provided | 2017-05-31 | criteria provided, single submitter | clinical testing | The D58G variant in the HADHB gene has not been reported previously as a pathogenic variant, nor as a benign variant, to our knowledge. The D58G variant is observed in 2/16512 (0.012%) alleles from individuals of South Asian background and in 2/66736 (0.003%) alleles from individuals of European background, in the ExAC dataset (Lek et al., 2016). The D58G variant is a non-conservative amino acid substitution, which is likely to impact secondary protein structure as these residues differ in polarity, charge, size and/or other properties. This substitution occurs at a position where amino acids with similar properties to Aspartic acid are tolerated across species. In silico analysis predicts this variant is probably damaging to the protein structure/function. We interpret D58G as a variant of uncertain significance. |
Labcorp Genetics |
RCV001208691 | SCV001380094 | uncertain significance | Mitochondrial trifunctional protein deficiency | 2021-09-01 | criteria provided, single submitter | clinical testing | This sequence change replaces aspartic acid with glycine at codon 58 of the HADHB protein (p.Asp58Gly). The aspartic acid residue is moderately conserved and there is a moderate physicochemical difference between aspartic acid and glycine. This variant is present in population databases (rs746076418, ExAC 0.01%). This variant has not been reported in the literature in individuals affected with HADHB-related conditions. ClinVar contains an entry for this variant (Variation ID: 430537). 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: "Possibly Damaging"; Align-GVGD: "Class C0"). 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. |