Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001063995 | SCV001228867 | pathogenic | X-linked severe combined immunodeficiency | 2019-03-25 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. Loss-of-function variants in IL2RG are known to be pathogenic (PMID: 9058718, 10794430). This variant has not been reported in the literature in individuals with IL2RG-related conditions. This variant is not present in population databases (ExAC no frequency). This sequence change creates a premature translational stop signal (p.Val99Serfs*48) in the IL2RG gene. It is expected to result in an absent or disrupted protein product. |
Lifecell International Pvt. |
RCV001063995 | SCV003925473 | pathogenic | X-linked severe combined immunodeficiency | criteria provided, single submitter | clinical testing | A Hemizygote Frameshift variant c.294delA in Exon 3 of the IL2RG gene that results in the amino acid substitution p.Val99fs*48 was identified. The observed variant has a miniimum allele frequency of 00/00% in gnomAD exomes and genomes, respectively. The severity of the impact of this variant on the protein is high, based on the effect of the protein and REVEL score . Rare Exome Variant Ensemble Learner (REVEL) is an ensembl method for predicting the pathogenicity of missense variants based on a combination of scores from 13 individual tools: MutPred, FATHMM v2.3, VEST 3.0, PolyPhen-2, SIFT, PROVEAN, MutationAssessor, MutationTaster, LRT, GERP++, SiPhy, phyloP, and phastCons. The REVEL score for an individual missense variant can range from 0 to 1, with higher scores reflecting greater likelihood that the variant is disease-causing. ClinVar has also classified this variant as Pathogenic( variant ID 858175). This variant has been observed in many individuals affected with Severe combined immunodeficiency, X-linked reported by (Niemela JE, et al., 2000). Based on the above evidence this variant has been classified as Pathogenic according to the ACMG guidelines. |