Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV005415447 | SCV006082471 | uncertain significance | Mitochondrial disease | 2024-11-11 | reviewed by expert panel | curation | The m.8319A>G variant in MT-TK has been reported in one individual with primary mitochondrial disease to date (PMID: 23463613; also likely same case reported in PMID: 31965079), in a person with Kearns Sayre syndrome (KSS) with the variant present at 87% in muscle. The variant was undetectable in blood from the healthy mother (PM6_supporting). There are no reports of additional families with this variant segregating with clinical manifestations. This variant is absent in the GenBank dataset, Helix dataset, and gnomAD v3.1.2 (PM2_supporting). In silico predictors are conflicting as the computational predictor MitoTIP suggests this variant is possibly pathogenic (69.6 percentile) but HmtVAR predicts it to be likely benign with a score of 0.25. There are no cybrids, single fiber studies, or other functional assays reported on this variant. In summary, this variant meets criteria to be classified as uncertain significance for primary mitochondrial disease inherited in a mitochondrial manner. This classification was approved by the NICHD/NINDS U24 ClinGen Mitochondrial Disease Variant Curation Expert Panel on November 11, 2024. Mitochondrial DNA-specific ACMG/AMP criteria applied (PMID: 32906214): PM2_supporting, PM6_supporting. |
Wong Mito Lab, |
RCV000850944 | SCV000993178 | likely pathogenic | MELAS syndrome | 2019-07-12 | criteria provided, single submitter | clinical testing | The NC_012920.1:m.8319A>G variant in MT-TK gene is interpreted to be a Likely Pathogenic variant based on the modified ACMG guidelines (unpublished). This variant meets the following evidence codes reported in the guidelines: PM2, PM9, PP3, PP6 |
MGZ Medical Genetics Center | RCV002290478 | SCV002580749 | uncertain significance | Kearns-Sayre syndrome | 2022-02-25 | criteria provided, single submitter | clinical testing |