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T-box transcription factor 3 governs a transcriptional program for the function of the mouse atrioventricular conduction system

Rajiv A. Mohan, Fernanda M. Bosada, Jan Hendrik van Weerd, Karel van Duijvenboden, Jianan Wang, Mathilda T.M. Mommersteeg, Ingeborg B. Hooijkaas, Vincent Wakker, Corrie de Gier-de Vries, Ruben Coronel, Gerard J.J. Boink, Jeroen Bakkers, Phil Barnett, Bastiaan J. Boukens, Vincent M. Christoffels

2020Proceedings of the National Academy of Sciences43 citationsDOIOpen Access PDF

Abstract

Significance Genome-wide association studies have identified common noncoding variants associated with ECG parameters in the TBX3 locus, suggesting that a modest change in TBX3/Tbx3 expression level affects heart function. We characterized the morphology, physiology, and gene expression profile of the atrioventricular conduction system in a Tbx3 haploinsufficient mouse model. We identified Tbx3-dependent regulatory DNA elements active in the atrioventricular conduction system and validated the functionality of these elements in vivo. Using genome editing, we deleted a regulatory DNA element from the mouse genome and show it drives expression of Cacna1g in the cardiac conduction system. Our study unveils a Tbx3-dependent gene regulatory network in the AV conduction system that contributes to heart electrophysiology.

Topics & Concepts

Transcription factorFunction (biology)Electrical conduction system of the heartCell biologyTranscription (linguistics)BiologyPhysicsInternal medicineGeneticsMedicineElectrocardiographyGenePhilosophyLinguisticsCongenital heart defects researchRNA Research and SplicingRNA modifications and cancer
T-box transcription factor 3 governs a transcriptional program for the function of the mouse atrioventricular conduction system | Litcius