Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000578820 | SCV000680762 | pathogenic | not provided | 2017-11-27 | criteria provided, single submitter | clinical testing | The C434X variant in the THRB gene has been reported previously in an individual with resistance to thyroid hormone and severe intellectual disability (Behr et al., 1997). This variant is predicted to cause loss of normal protein function through protein truncation, as the last 28 amino acids are lost. Functional studies demonstrate a damaging effect with dominant negative activity, silencing of basal gene transcription, and abnormal DNA-binding properties (Behr et al., 1997). The C434X variant is not observed in large population cohorts (Lek et al., 2016). We interpret C434X as a pathogenic variant. |
OMIM | RCV000013393 | SCV000033640 | pathogenic | Thyroid hormone resistance, generalized, autosomal dominant | 1997-04-01 | no assertion criteria provided | literature only | |
Clinical Molecular Genetics Laboratory, |
RCV000013393 | SCV000692427 | pathogenic | Thyroid hormone resistance, generalized, autosomal dominant | 2011-05-05 | no assertion criteria provided | clinical testing |