Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV002230968 | SCV000629980 | pathogenic | Primary pulmonary hypertension | 2023-07-30 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. ClinVar contains an entry for this variant (Variation ID: 425685). This premature translational stop signal has been observed in individual(s) with pulmonary arterial hypertension (PMID: 18356561, 21801371, 26387786). This variant is not present in population databases (gnomAD no frequency). This sequence change creates a premature translational stop signal (p.Trp16*) in the BMPR2 gene. It is expected to result in an absent or disrupted protein product. Loss-of-function variants in BMPR2 are known to be pathogenic (PMID: 16429395). |
Genetics and Molecular Pathology, |
RCV000488825 | SCV002556829 | pathogenic | Pulmonary hypertension, primary, 1 | 2022-03-13 | criteria provided, single submitter | clinical testing | |
Rare Disease Genomics Group, |
RCV000488825 | SCV000575957 | pathogenic | Pulmonary hypertension, primary, 1 | no assertion criteria provided | literature only | ||
NIHR Bioresource Rare Diseases, |
RCV001003644 | SCV001162087 | pathogenic | Pulmonary arterial hypertension | no assertion criteria provided | research | ||
Prevention |
RCV004551606 | SCV004115800 | pathogenic | BMPR2-related disorder | 2024-02-07 | no assertion criteria provided | clinical testing | The BMPR2 c.48G>A variant is predicted to result in premature protein termination (p.Trp16*). This variant has been reported in multiple individuals with idiopathic or hereditary pulmonary arterial hypertension (Sztrymf et al. 2008. PubMed ID: 18356561; Dewachter et al. 2009. PubMed ID: 19324947; Liu et al. 2011. PubMed ID: 21737554; Pfarr et al. 2011. PubMed ID: 21801371; Machado et al. 2015. PubMed ID: 26387786; Table S1, Eichstaedt et al. 2022. PubMed ID: 35346192). This variant has not been reported in a large population database, indicating this variant is rare. Nonsense variants in BMPR2 are expected to be pathogenic. This variant is interpreted as pathogenic. |