Abstract
Many of the major discoveries in the fields of genetics and developmental biology have been made using the fruit fly, Drosophila melanogaster. With regard to heart development, the conserved network of core cardiac transcription factors that underlies cardiogenesis has been studied in great detail in the fly, and the importance of several signaling pathways that regulate heart morphogenesis, such as Slit/Robo, was first shown in the fly model. Recent technological advances have led to a large increase in the genomic data available from patients with congenital heart disease (CHD). This has highlighted a number of candidate genes and gene networks that are potentially involved in CHD. To validate genes and genetic interactions among candidate CHD-causing alleles and to better understand heart formation in general are major tasks. The specific limitations of the various cardiac model systems currently employed (mammalian and fish models) provide a niche for the fly model, despite its evolutionary distance to vertebrates and humans. Here, we review recent advances made using the Drosophila embryo that identify factors relevant for heart formation. These underline how this model organism still is invaluable for a better understanding of CHD.
Highlights
Congenital heart disease (CHD) is the most frequently diagnosed birth defect, with an incident rate of about 1% among newborns [1,2]
Current vertebrate models of heart development are mouse, chicken and zebrafish embryos, which are complemented by cell-based assays, including heart tissue engineered from induced pluripotent stem cells [9]
HAND [33], Neuromancer1/2-TBX20 [34,35], Tail-up/Islet-1 [36,37] and the COUP transcription factor, Seven-up [38]. This genetic and mutational interrogation of cardiogenesis was recently complemented by ChIP-on-chip studies of whole embryos [39,40], which globally identified the transcriptional activity and regulatory networks controlled by Tinman and several other cardiac transcription factors
Summary
Congenital heart disease (CHD) is the most frequently diagnosed birth defect, with an incident rate of about 1% among newborns [1,2]. Current vertebrate models of heart development are mouse, chicken and zebrafish embryos (for reviews, see [5,6,7,8]), which are complemented by cell-based assays, including heart tissue engineered from induced pluripotent stem cells (iPSCs) [9]. These models established valuable concepts for the number and origin of cardiac progenitor cells, lineage restriction and the respective contributions of these cells to different regions of the heart. We review recent advances in the understanding of the molecular-genetic control of heart morphogenesis in Drosophila
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