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Genome-Wide Analysis of Facial Regionalization in Zebrafish

Authors :
Maxwell Bay
Michael A. Bonaguidi
Pengfei Xu
Amjad Askary
Bartosz Balczerski
J. Gage Crump
Lindsey Barske
Paul A. Bump
Publication Year :
2017
Publisher :
Cold Spring Harbor Laboratory, 2017.

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) andemx2, 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 StatementUsing 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.

Details

Database :
OpenAIRE
Accession number :
edsair.doi.dedup.....33c2cd8a98144db097f213c938905b97
Full Text :
https://doi.org/10.1101/114801