Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Mayo Clinic Laboratories, |
RCV000660521 | SCV000782622 | pathogenic | not provided | 2017-01-18 | criteria provided, single submitter | clinical testing | |
Wendy Chung Laboratory, |
RCV000664178 | SCV000784737 | uncertain significance | Pulmonary arterial hypertension associated with congenital heart disease | 2018-06-27 | criteria provided, single submitter | case-control | |
Wendy Chung Laboratory, |
RCV001829824 | SCV002097214 | likely pathogenic | Pulmonary hypertension, primary, 1 | 2019-11-01 | criteria provided, single submitter | research | |
Labcorp Genetics |
RCV000660521 | SCV002122667 | pathogenic | not provided | 2023-04-09 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. ClinVar contains an entry for this variant (Variation ID: 547941). Disruption of this splice site has been observed in individuals with pulmonary arterial hypertension (PMID: 29631995, 30029678). This variant is not present in population databases (gnomAD no frequency). This sequence change affects a donor splice site in intron 5 of the TBX4 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 TBX4 are known to be pathogenic (PMID: 15106123, 31151956, 31761294, 31965066, 32079640). |
Prevention |
RCV004547833 | SCV004102905 | pathogenic | TBX4-related disorder | 2023-09-15 | criteria provided, single submitter | clinical testing | The TBX4 c.702+1G>A variant is predicted to disrupt the GT donor site and interfere with normal splicing. This variant has been reported in individuals with pulmonary arterial hypertension and/or small patella syndrome (Table S1, Zhu et al. 2018. PubMed ID: 29631995; Table S2, Zhu et al. 2018. PubMed ID: 30029678; Table S2, Klee et al. 2020. PubMed ID: 33144682). This variant has not been reported in a large population database (http://gnomad.broadinstitute.org), indicating this variant is rare. Variants that disrupt the consensus splice donor site in TBX4 are expected to be pathogenic. This variant is interpreted as pathogenic. |