PUBLICATION
Genome-wide analysis of facial skeletal regionalization in zebrafish
- Authors
- Askary, A., Xu, P., Barske, L., Bay, M., Bump, P., Balczerski, B., Bonaguidi, M.A., Crump, J.G.
- ID
- ZDB-PUB-170715-7
- Date
- 2017
- Source
- Development (Cambridge, England) 144(16): 2994-3005 (Journal)
- Registered Authors
- Balczerski, Bartosz, Barske, Lindsey, Crump, Gage DeKoeyer, Xu, Peng-Fei
- Keywords
- Bone, Cartilage, Cranial neural crest, Craniofacial, Dlx5a, Dorsoventral patterning, Edn1, Emx2, Endothelin, Fsta, Hand2, Jagged-Notch, Jaw, Mrrf, Skeleton, Zebrafish
- Datasets
- GEO:GSE95812
- MeSH Terms
-
- Flow Cytometry
- In Situ Hybridization
- Endothelin-1/genetics
- Endothelin-1/metabolism
- Branchial Region/embryology
- Branchial Region/metabolism
- Gene Expression Regulation, Developmental/genetics
- Gene Expression Regulation, Developmental/physiology
- Body Patterning/genetics
- Body Patterning/physiology
- Zebrafish/embryology*
- Zebrafish/genetics*
- Animals
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 28705894 Full text @ Development
Citation
Askary, A., Xu, P., Barske, L., Bay, M., Bump, P., Balczerski, B., Bonaguidi, M.A., Crump, J.G. (2017) Genome-wide analysis of facial skeletal regionalization in zebrafish. Development (Cambridge, England). 144(16):2994-3005.
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 Endothelin 1 (Edn1) signaling in the intermediate joint-forming region, yet a surprisingly minor role in ventralmost regions. Analysis of co-variance across multiple sequencing experiments further reveals clusters of co-regulated genes, with in situ hybridization confirming the domain-specific expression of novel genes. We then created loss-of-function alleles for 12 genes and 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 Edn1 and points to novel candidates for craniofacial disorders.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping