Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000493155 | SCV000582547 | pathogenic | not provided | 2015-10-13 | criteria provided, single submitter | clinical testing | The Y168S pathogenic variant in the GAMT gene has been previously reported in the homozygous state in an individual with phenotypic and biochemical evidence of GAMT deficiency (Bodamer et al., 2009). It was not observed in approximately 6,500 individuals of European and African American ancestry in the NHLBI Exome Sequencing Project, indicating it is not a common benign variant in these populations. This semi-conservative substitution occurs at a position that is conserved across species, and in silico analysis predicts this variant is probably damaging to the protein structure/function. Additionally, missense variants in nearby residues (C169R/Y, L166P) have been reported in the Human Gene Mutation Database in association with GAMT deficiency (Stenson et al., 2014), supporting the functional importance of this region of the protein. Therefore, Y168S is considered a pathogenic variant. |
ARUP Laboratories, |
RCV000493155 | SCV000883935 | likely pathogenic | not provided | 2017-12-05 | criteria provided, single submitter | clinical testing |