Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000004285 | SCV004298068 | pathogenic | Aicardi-Goutieres syndrome 5 | 2023-01-17 | criteria provided, single submitter | clinical testing | ClinVar contains an entry for this variant (Variation ID: 4070). This premature translational stop signal has been observed in individual(s) with Aicardi-Goutieres syndrome (PMID: 19525956). This variant is not present in population databases (gnomAD no frequency). This sequence change creates a premature translational stop signal (p.Gln149*) in the SAMHD1 gene. It is expected to result in an absent or disrupted protein product. Loss-of-function variants in SAMHD1 are known to be pathogenic (PMID: 19525956, 22461318). Algorithms developed to predict the effect of variants on protein structure and function are not available or were not evaluated for this variant. For these reasons, this variant has been classified as Pathogenic. Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. Experimental studies have shown that this premature translational stop signal affects SAMHD1 function (PMID: 19525956, 22461318). |
OMIM | RCV000004285 | SCV000024451 | pathogenic | Aicardi-Goutieres syndrome 5 | 2009-07-01 | no assertion criteria provided | literature only | |
Gene |
RCV000004285 | SCV000147934 | not provided | Aicardi-Goutieres syndrome 5 | no assertion provided | literature only |