Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Breda Genetics srl | RCV001328473 | SCV001519520 | uncertain significance | Developmental and epileptic encephalopathy, 64 | 2020-10-23 | criteria provided, single submitter | clinical testing | The variant c.274C>T (p.Arg92*) in the RHOBTB2 is predicted to create a premature stop codon at amino acid position Arg92, which is likely to result in a truncated protein or protein loss due to nonsense-mediated messenger decay (NMD). There is no information on frequency in gnomAD, 1000 Genomes or NHLI Exome Sequencing Project (ESP). It is important to highlight that only missense variants have been reported as pathogenic so far, as RHOBTB2 mutations seem to result more likely in altered protein function rather than haploinsufficiency or a loss of function (Straub et al., 2018, PMID: 29276004). Therefore, despite its predicted impact as nonsense mutation, we interpret this variant as of uncertain significance, without excluding the possibility that it’s actually a rare benign variant. |
Ce |
RCV003438742 | SCV004164488 | uncertain significance | not provided | 2022-03-01 | criteria provided, single submitter | clinical testing | RHOBTB2: PM2 |
Labcorp Genetics |
RCV003438742 | SCV004484601 | uncertain significance | not provided | 2023-02-08 | criteria provided, single submitter | clinical testing | This sequence change creates a premature translational stop signal (p.Arg92*) in the RHOBTB2 gene. It is expected to result in an absent or disrupted protein product. However, the current clinical and genetic evidence is not sufficient to establish whether loss-of-function variants in RHOBTB2 cause disease. This variant is not present in population databases (gnomAD no frequency). This variant has not been reported in the literature in individuals affected with RHOBTB2-related conditions. ClinVar contains an entry for this variant (Variation ID: 1027644). In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. |