PUBLICATION

Disruption of pdgfra alters endocardial and myocardial fusion during zebrafish cardiac assembly.

Authors
El-Rass, S., Eisa-Beygi, S., Khong, E., Brand-Arzamendi, K., Mauro, A., Zhang, H., Clark, K.J., Ekker, S.C., Wen, X.Y.
ID
ZDB-PUB-170209-18
Date
2017
Source
Biology Open   6(3): 348-357 (Journal)
Registered Authors
Clark, Karl, Eisa-Beygi, Shahram, Ekker, Stephen C., Wen, Xiao-Yan
Keywords
Cardiac fusion, Gene trapping, Heart development, Pdgfra, Zebrafish
MeSH Terms
none
PubMed
28167492 Full text @ Biol. Open
Abstract
Cardiac development in vertebrates is a finely tuned process regulated by a set of conserved signaling pathways. Perturbations of these processes are often associated with congenital cardiac malformations. Platelet-derived growth factor receptor α (PDGFRα) is a highly conserved tyrosine kinase receptor, which is essential for development and organogenesis. Disruption of Pdgfrα function in murine models is embryonic lethal due to severe cardiovascular defects, suggesting a role in cardiac development, thus necessitating the use of alternative models to explore its precise function. In this study, we generated a zebrafish pdgfra mutant line by gene trapping, in which the Pdgfra protein is truncated and fused with mRFP (Pdgfra-mRFP). Our results demonstrate that pdgfra mutants have defects in cardiac morphology as a result of abnormal fusion of myocardial precursors. Expression analysis of the developing heart at later stages suggested that Pdgfra-mRFP is expressed in the endocardium. Further examination of the endocardium in pdgfra mutants revealed defective endocardial migration to the midline, where cardiac fusion eventually occurs. Together, our data suggests that pdgfra is required for proper medial migration of both endocardial and myocardial precursors, an essential step required for cardiac assembly and development.
Genes / Markers
Figures
Figure Gallery (12 images) / 2
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
pdgfrasmh1300Gt/+ (TU)controlDay 4
pdgfrasmh1300Gt/smh1300Gt (TU)control14-19 somites to Prim-5
pdgfrasmh1300Gt/smh1300Gt (TU)control14-19 somites to Prim-5
pdgfrasmh1300Gt/smh1300Gt (TU)control14-19 somites to Prim-5
pdgfrasmh1300Gt/smh1300Gt (TU)control20-25 somites
pdgfrasmh1300Gt/smh1300Gt (TU)control20-25 somites
pdgfrasmh1300Gt/smh1300Gt (TU)control20-25 somites
pdgfrasmh1300Gt/smh1300Gt (TU)control20-25 somites
pdgfrasmh1300Gt/smh1300Gt (TU)control20-25 somites to Prim-5
pdgfrasmh1300Gt/smh1300Gt (TU)control26+ somites to Prim-5
1 - 10 of 35
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
ba2TgTransgenic Insertion
    s843TgTransgenic Insertion
      smh1300GtTransgenic Insertion
      twu34TgTransgenic Insertion
        1 - 4 of 4
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        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        No data available
        Fish
        Fish
        ba2Tg
        pdgfrasmh1300Gt/smh1300Gt; ba2Tg
        pdgfrasmh1300Gt/smh1300Gt (TU)
        pdgfrasmh1300Gt/smh1300Gt; twu34Tg
        pdgfrasmh1300Gt/+; s843Tg
        pdgfrasmh1300Gt/+ (TU)
        pdgfrasmh1300Gt/+; twu34Tg
        s843Tg
        TU
        twu34Tg
        1 - 10 of 10
        Show
        Antibodies
        No data available
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        EGFPEFGEGFP
        mRFP1EFGmRFP1
        1 - 2 of 2
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        Mapping
        No data available
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        Citation
        El-Rass, S., Eisa-Beygi, S., Khong, E., Brand-Arzamendi, K., Mauro, A., Zhang, H., Clark, K.J., Ekker, S.C., Wen, X.Y. (2017) Disruption of pdgfra alters endocardial and myocardial fusion during zebrafish cardiac assembly.. Biology Open. 6(3):348-357.