Total submissions: 7
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
NIHR Bioresource Rare Diseases, |
RCV001027614 | SCV001190186 | likely pathogenic | Inherited Immunodeficiency Diseases | 2019-01-01 | criteria provided, single submitter | research | |
Ce |
RCV001091955 | SCV001248258 | pathogenic | not provided | 2019-06-01 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV001389160 | SCV001590426 | pathogenic | Combined immunodeficiency with skin granulomas; Severe combined immunodeficiency, autosomal recessive, T cell-negative, B cell-negative, NK cell-positive | 2024-05-22 | criteria provided, single submitter | clinical testing | This sequence change replaces arginine, which is basic and polar, with histidine, which is basic and polar, at codon 737 of the RAG1 protein (p.Arg737His). This variant is present in population databases (rs104894286, gnomAD 0.02%). This missense change has been observed in individual(s) with severe combined immunodeficiency and Omenn syndrome (PMID: 9630231, 17572155, 28109013, 30290665, 30858051). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. This variant is also known as G2322A and c.2322G>A. ClinVar contains an entry for this variant (Variation ID: 13149). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) has been performed at Invitae for this missense variant, however the output from this modeling did not meet the statistical confidence thresholds required to predict the impact of this variant on RAG1 protein function. Experimental studies have shown that this missense change affects RAG1 function (PMID: 9630231). For these reasons, this variant has been classified as Pathogenic. |
Genomic Medicine Center of Excellence, |
RCV003227600 | SCV003924311 | pathogenic | Severe combined immunodeficiency, autosomal recessive, T cell-negative, B cell-negative, NK cell-positive | 2023-05-08 | criteria provided, single submitter | research | |
Baylor Genetics | RCV003473093 | SCV004200479 | pathogenic | Combined immunodeficiency due to partial RAG1 deficiency | 2024-03-27 | criteria provided, single submitter | clinical testing | |
Fulgent Genetics, |
RCV005042043 | SCV005683483 | pathogenic | Combined immunodeficiency due to partial RAG1 deficiency; Combined immunodeficiency with skin granulomas; Histiocytic medullary reticulosis; Severe combined immunodeficiency, autosomal recessive, T cell-negative, B cell-negative, NK cell-positive | 2024-06-25 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000014032 | SCV000034279 | pathogenic | Histiocytic medullary reticulosis | 1998-05-29 | no assertion criteria provided | literature only |