TY - JOUR T1 - Loss of embryonic neural crest cardiomyocytes causes adult hypertrophic cardiomyopathy JF - bioRxiv DO - 10.1101/270652 SP - 270652 AU - Sarah Abdul-Wajid AU - Bradley L Demarest AU - H Joseph Yost Y1 - 2018/01/01 UR - http://biorxiv.org/content/early/2018/02/23/270652.abstract N2 - Neural crest cells migrate to the embryonic heart and transform into a small number of cardiomyocytes, but their functions in the developing and adult heart are unknown. Here, we map the fates of neural crest derived cardiomyocytes (NC-Cms) and genetically ablate them in embryogenesis in zebrafish. Specific NC-Cm ablation results in aberrant trabeculation patterns and altered Notch signaling, but is not detrimental to the development of the fish or early heart function. Strikingly, embryonic NC-Cm ablation results in adult fish that show severely hypertrabeculated hearts, altered cardiomyocyte size, diminished adult heart capacity and consequently poor physiological response to cardiac stress tests. Thus, we identify a novel developmental mechanism and genetic pathway that predisposes adults to hypertrophic cardiomyopathy and provides the first zebrafish model of adult-onset heart failure. ER -