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Genome-Wide Analysis of Facial Regionalization in Zebrafish

Amjad Askary, Pengfei Xu, Lindsey Barske, Maxwell Bay, Paul Bump, Bartosz Balczerski, Michael A. Bonaguidi, J. Gage Crump
doi: https://doi.org/10.1101/114801
Amjad Askary
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Pengfei Xu
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Lindsey Barske
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Maxwell Bay
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Paul Bump
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Bartosz Balczerski
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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Michael A. Bonaguidi
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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J. Gage Crump
Eli and Edythe Broad California Institute of Regenerative Medicine Center, Department of Stem Cell Biology and Regenerative Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
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  • For correspondence: gcrump@usc.edu
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Abstract

Patterning of the facial skeleton involves the precise deployment of thousands of genes in distinct regions of the pharyngeal arches. Despite the significance for craniofacial development, how genetic programs drive this regionalization remains incompletely understood. Here we use combinatorial labeling of zebrafish cranial neural crest-derived cells (CNCCs) to define global gene expression along the dorsoventral axis of the developing arches. Intersection of region-specific transcriptomes with expression changes in response to signaling perturbations demonstrates complex roles for Endothelin1 (Edn1) signaling in the intermediate joint-forming region yet a surprisingly minor role in ventral-most regions. Analysis of co-variance across multiple sequencing experiments further reveals clusters of coregulated genes, with in situ hybridization confirming the domain-specific expression of novel genes. We then performed mutational analysis of a number of these genes, which uncovered antagonistic functions of two new Edn1 targets, follistatin a (fsta) and emx2, in regulating cartilaginous joints in the hyoid arch. Our unbiased discovery and functional analysis of genes with regional expression in zebrafish arch CNCCs reveals complex regulation by Ednl and points to novel candidates for craniofacial disorders.

Summary Statement Using zebrafish to purify distinct groups of embryonic cells, Askary et al. have created a detailed map of how thousands of genes are deployed to shape the developing face.

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  • ↵1 Co-first authors

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The copyright holder for this preprint is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. All rights reserved. No reuse allowed without permission.
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Posted March 07, 2017.
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Genome-Wide Analysis of Facial Regionalization in Zebrafish
Amjad Askary, Pengfei Xu, Lindsey Barske, Maxwell Bay, Paul Bump, Bartosz Balczerski, Michael A. Bonaguidi, J. Gage Crump
bioRxiv 114801; doi: https://doi.org/10.1101/114801
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Genome-Wide Analysis of Facial Regionalization in Zebrafish
Amjad Askary, Pengfei Xu, Lindsey Barske, Maxwell Bay, Paul Bump, Bartosz Balczerski, Michael A. Bonaguidi, J. Gage Crump
bioRxiv 114801; doi: https://doi.org/10.1101/114801

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