Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001969612 | SCV002266196 | likely pathogenic | Hyper-IgM syndrome type 2 | 2021-07-15 | criteria provided, single submitter | clinical testing | This sequence change affects an acceptor splice site in intron 4 of the AICDA gene. While this variant is not anticipated to result in nonsense mediated decay, it likely alters RNA splicing and results in a disrupted protein product. In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. Nucleotide substitutions within the consensus splice site are a relatively common cause of aberrant splicing (PMID: 17576681, 9536098). 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 disruption of this splice site affects AICDA protein function (PMID: 12910268). Algorithms developed to predict the effect of variants on protein structure and function are not available or were not evaluated for this variant. This variant is also known as V5-2A>G. Disruption of this splice site has been observed in individuals with autosomal recessive hyper IgM syndrome (PMID: 14962793; Invitae). This variant is present in population databases (rs772388034, ExAC 0.006%). |