Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001220239 | SCV001392219 | pathogenic | Mitochondrial trifunctional protein deficiency; Long chain 3-hydroxyacyl-CoA dehydrogenase deficiency | 2024-04-15 | criteria provided, single submitter | clinical testing | This sequence change replaces arginine, which is basic and polar, with histidine, which is basic and polar, at codon 676 of the HADHA protein (p.Arg676His). This variant is present in population databases (no rsID available, gnomAD 0.007%). This missense change has been observed in individual(s) with HADHA-related conditions (PMID: 10352164, 21549624, 26109258; Invitae). This variant is also known as Arg640His. ClinVar contains an entry for this variant (Variation ID: 948895). 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 HADHA protein function with a positive predictive value of 80%. This variant disrupts the p.Arg676 amino acid residue in HADHA. Other variant(s) that disrupt this residue have been observed in individuals with HADHA-related conditions (PMID: 24305961), which suggests that this may be a clinically significant amino acid residue. For these reasons, this variant has been classified as Pathogenic. |
Gene |
RCV001587243 | SCV001826883 | likely pathogenic | not provided | 2019-10-14 | criteria provided, single submitter | clinical testing | In silico analysis, which includes protein predictors and evolutionary conservation, supports a deleterious effect; This variant is associated with the following publications: (PMID: 29915090, 15902556, 21549624, 10352164) |
Baylor Genetics | RCV003469379 | SCV004191792 | likely pathogenic | Long chain 3-hydroxyacyl-CoA dehydrogenase deficiency | 2024-03-02 | criteria provided, single submitter | clinical testing | |
Fulgent Genetics, |
RCV005021523 | SCV005651669 | likely pathogenic | Long chain 3-hydroxyacyl-CoA dehydrogenase deficiency; Mitochondrial trifunctional protein deficiency 1 | 2024-04-03 | criteria provided, single submitter | clinical testing |