Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Victorian Clinical Genetics Services, |
RCV000010628 | SCV002558056 | uncertain significance | Glycogen storage disease due to phosphoglycerate kinase 1 deficiency | 2022-02-02 | criteria provided, single submitter | clinical testing | Based on the classification scheme VCGS_Germline_v1.3.4, this variant is classified as VUS-3A. Following criteria are met: 0102 - Loss of function is a known mechanism of disease in this gene and is associated with phosphoglycerate kinase 1 deficiency (MIM#30065). (I) 0109 - This gene is associated with X-linked recessive disease. (I) 0115 - Variants in this gene are known to have variable expressivity. Affected individuals have a highly variable phenotype and age of onset (OMIM). (I) 0200 - Variant is predicted to result in a missense amino acid change from isoleucine to threonine. (I) 0251 - This variant is heterozygous. (I) 0304 - Variant is present in gnomAD <0.01 for a recessive condition (v2) (0 heterozygotes, 0 homozygotes, 2 hemizygotes). (SP) 0501 - Missense variant consistently predicted to be damaging by multiple in silico tools or highly conserved with a major amino acid change. (SP) 0600 - Variant is located in the annotated PGK domain (Pfam). (I) 0705 - No comparable missense variants have previous evidence for pathogenicity. (I) 0802 - This variant has moderate previous evidence of pathogenicity in unrelated individuals. This variant has been reported in 3 patients with phosphoglycerate kinase 1 deficiency (ClinVar, PMIDs: 2715616, 27848944). (SP) 0905 - No published segregation evidence has been identified for this variant. (I) 1010 - Functional evidence for this variant is inconclusive. This variant was shown to faintly affect molecular properties and acted similar to wild-type, however these results correlate with the wide spectrum of clinical symptoms (PMID: 22348148). (I) Legend: (SP) - Supporting pathogenic, (I) - Information, (SB) - Supporting benign |
OMIM | RCV000010628 | SCV000030854 | pathogenic | Glycogen storage disease due to phosphoglycerate kinase 1 deficiency | 2006-06-01 | no assertion criteria provided | literature only | |
Biochemical Molecular Genetic Laboratory, |
RCV000010628 | SCV001133217 | likely pathogenic | Glycogen storage disease due to phosphoglycerate kinase 1 deficiency | 2019-09-26 | no assertion criteria provided | clinical testing |