Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV000767984 | SCV005619908 | likely benign | Deficiency of guanidinoacetate methyltransferase | 2024-09-11 | reviewed by expert panel | curation | The NM_000156.6:c.570+161T>A variant in GAMT is an intronic variant affecting a nucleotide within the consensus splice site of intron 5. To our knowledge, this variant has not been reported in the literature and results of functional studies are unavailable. The highest population frequency in gnomAD v4.1.0. is 0.001162 (85/73150 alleles; no homozygotes) in the African / African American population, which is higher than the ClinGen CCDS VCEP’s threshold for BS1 (>0.001), and therefore meets this criterion (BS1). The computational splicing predictor SpliceAI gives a score of 0.0 for donor loss and suggests that the variant has no impact on splicing (BP4). There is a ClinVar entry for this variant (Variation ID: 625952). In summary, this variant meets the criteria to be classified as likely benign for GAMT deficiency. GAMT-specific ACMG/AMP codes met, as specified by the ClinGen Cerebral Creatine Deficiency Syndromes VCEP (Specifications Version 2.0.0): BS1, BP4. (Classification approved by the ClinGen Creatine Deficiency Syndromes Variant Curation Expert Panel on September 11, 2024). |
Center for Genomics, |
RCV000767984 | SCV000898727 | uncertain significance | Deficiency of guanidinoacetate methyltransferase | 2021-03-30 | criteria provided, single submitter | clinical testing | GAMT NM_138924 exon5 p.Leu244Gln (c.731T>A): This variant has not been reported in the literature but is present in 0.1% (25/16114) of Ashkenazi Jewish individuals in the Genome Aggregation Database (http://gnomad.broadinstitute.org/rs368296913). Evolutionary conservation and computational predictive tools for this variant are limited or unavailable. In summary, data on this variant is insufficient for disease classification. Therefore, the clinical significance of this variant is uncertain. |
Prevention |
RCV003955495 | SCV004776094 | likely benign | GAMT-related disorder | 2022-02-23 | no assertion criteria provided | clinical testing | This variant is classified as likely benign based on ACMG/AMP sequence variant interpretation guidelines (Richards et al. 2015 PMID: 25741868, with internal and published modifications). |