Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Women's Health and Genetics/Laboratory Corporation of America, |
RCV000030053 | SCV000052708 | likely pathogenic | X-linked severe combined immunodeficiency | 2011-08-18 | criteria provided, single submitter | curation | Converted during submission to Likely pathogenic. |
Gene |
RCV000430650 | SCV000516115 | pathogenic | not provided | 2015-03-30 | criteria provided, single submitter | clinical testing | The c.270-1 G>T splice site variant in the IL2RG gene destroys the canonical spliceacceptor site in intron 2. It is predicted to cause abnormal gene splicing, either leading to an abnormal messagethat is subject to nonsense-mediated mRNA decay, or to an abnormal protein product if the message is usedfor protein translation. The c.270-1 G>T variant was not observed in approximately 6,500 individuals ofEuropean and African American ancestry in the NHLBI Exome Sequencing Project, indicating it is not acommon benign variant in these populations. We interpret the c.270-1 G>T variant as pathogenic. |
Invitae | RCV000030053 | SCV002157078 | pathogenic | X-linked severe combined immunodeficiency | 2021-03-22 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. Experimental studies have shown that disruption of this splice site results in altered mRNA splicing and is expected to lead to the loss of protein expression (PMID: 28359783). Disruption of this splice site ‚Äãhas been observed in individual(s) with severe combined immunodeficiency (PMID: 28359783, Invitae). ClinVar contains an entry for this variant (Variation ID: 36384) This variant is not present in population databases (ExAC no frequency). This sequence change affects an acceptor splice site in intron 2 of the IL2RG gene. It is expected to disrupt RNA splicing. Variants that disrupt the donor or acceptor splice site typically lead to a loss of protein function (PMID: 16199547), and loss-of-function variants in IL2RG are known to be pathogenic (PMID: 9058718, 10794430). |