Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
SIB Swiss Institute of Bioinformatics | RCV002254491 | SCV002525747 | uncertain significance | Combined oxidative phosphorylation defect type 24 | 2022-06-08 | criteria provided, single submitter | curation | This variant is interpreted as a variant of uncertain significance for Combined oxidative phosphorylation deficiency 24, autosomal recessive. The following ACMG Tag(s) were applied: Absent from controls (or at extremely low frequency if recessive) in Exome Sequencing Project, 1000 Genomes Project, or Exome Aggregation Consortium (PM2); Multiple lines of computational evidence support a deleterious effect on the gene or gene product (PP3); Novel missense change at an amino acid residue where a different missense change determined to be pathogenic has been seen before (PM5). |
Illumina Laboratory Services, |
RCV003126207 | SCV003802787 | likely pathogenic | not provided | 2022-09-28 | criteria provided, single submitter | clinical testing | The NARS2 c.1141A>G (p.Asn381Asp) missense variant results in the substitution of asparagine at amino acid position 381 with aspartic acid. This variant has been reported in a compound heterozygous state in one individual with combined oxidative phosphorylation deficiency (COXPD) (PMID: 35558980). Another variant at the same amino acid position, c.1142A>G (p.Asn381Ser), has been reported in a compound heterozygous state in one individual with COXPD (PMID: 25807530). The c.1141A>G variant is not found in version 2.1.1 or version 3.1.2 of the Genome Aggregation Database. Western blot analysis demonstrated the variant showed lower steady-state levels compared to that of the wild type (PMID: 35558980). The c.1141A>G variant lies within the catalytic domain of the protein. Based on the available evidence, the c.1141A>G (p.Asn381Asp) variant is classified as likely pathogenic for primary mitochondrial disease. |