Is Essential for Cardiac Development and Function.

Cardiovascular diseases (CVDs) are the prevalent cause of mortality worldwide. A combination of environmental and genetic effectors modulates the risk of developing them. Thus, it is vital to identify candidate genes and elucidate their role in the manifestation of the disease. Large-scale human studies have revealed the implication of Craniofacial Development Protein 1 (CFDP1) in Coronary Artery Disease (CAD). CFDP1 belongs to the evolutionary conserved Bucentaur (BCNT) family, and to date, its function and mechanism of action in Cardiovascular Development are still unclear. We utilized zebrafish to investigate the role of in the developing heart due to the high genomic homology, similarity in heart physiology, and ease of experimental manipulations. We showed that was expressed during development, and we tested two morpholinos and generated a mutant line. The embryos developed arrhythmic hearts and exhibited defective cardiac performance, which led to a lethal phenotype. Findings from both knockdown and knockout experiments showed that abrogation of leads to downregulation of Wnt signaling in embryonic hearts during valve development but without affecting Notch activation in this process. The zebrafish mutant line provides a valuable tool for unveiling the novel mechanism of regulating cardiac physiology and function. is essential for cardiac development, a previously unreported phenotype most likely due to early lethality in mice. The detected phenotype of bradycardia and arrhythmias is an observation with potential clinical relevance for humans carrying heterozygous CFDP1 mutations and their risk of developing CAD.