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- W2004320797 abstract "The T-box transcription factor Tbx5 (Tbx5a in zebrafish) plays a crucial role in the formation of cardiac chambers in a dose-dependent manner. Its deregulation leads to congenital heart disease. However, little is known regarding its regulation. Here we isolate a zebrafish mutant with heart malformations, called 34c. The affected gene is identified as kctd10, a member of the potassium channel tetramerization domain (KCTD)-containing family. In the mutant, the expressions of the atrioventricular canal marker genes, such as tbx2b, hyaluronan synthase 2 (has2), notch1b and bmp4, are changed. The knockdown of tbx5 rescues the ectopic expression of has2, and knockdown of either tbx5a or has2 alleviates the heart defects. We show that Kctd10 directly binds to Tbx5 to repress its transcriptional activity. Our results reveal a new essential factor for cardiac development and suggest that KCTD10 could be considered as a new causative gene of congenital heart disease. T-box transcription factors such as Tbx5 have essential roles during cardiac development. Here the authors show that a member of the potassium channel tetramerization domain-containing family, Kctd10 is required for zebrafish heart development and represses the transcriptional activity of Tbx5." @default.
- W2004320797 created "2016-06-24" @default.
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- W2004320797 date "2014-01-16" @default.
- W2004320797 modified "2023-10-16" @default.
- W2004320797 title "Kctd10 regulates heart morphogenesis by repressing the transcriptional activity of Tbx5a in zebrafish" @default.
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- W2004320797 doi "https://doi.org/10.1038/ncomms4153" @default.
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