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
  • In Situ Hybridization
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Flow Cytometry
  • Animals
  • Body Patterning/genetics
  • Body Patterning/physiology
  • Branchial Region/embryology
  • Branchial Region/metabolism
  • Zebrafish/embryology*
  • Zebrafish/genetics*
  • Gene Expression Regulation, Developmental/genetics
  • Gene Expression Regulation, Developmental/physiology
  • Endothelin-1/genetics
  • Endothelin-1/metabolism
(all 15)
PubMed
28705894 Full text @ Development
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
Figures
Figure Gallery (8 images)
Show all Figures
Expression
Phenotype
Fish Conditions Stage Phenotype Figure
ccn2ael654/el654standard conditionsDay 5
ccn2bel689/el689standard conditionsDay 5
cd248ael679/el679standard conditionsDay 5
cdh11el520/el520standard conditionsDay 5
edn1tf216b/tf216bstandard conditionsDay 5
edn1tf216b/tf216b; el10Tg; y1Tgstandard conditionsPrim-25
edn1tf216b/tf216b; el10Tg; y1Tgstandard conditionsPrim-25
edn1tf216b/tf216b; el10Tg; y1Tgstandard conditionsPrim-25
edn1tf216b/tf216b; el10Tg; y1Tgstandard conditionsPrim-25
edn1tf216b/tf216b; el10Tg; y1Tgstandard conditionsPrim-25
1 - 10 of 75
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
b1105
    		Point Mutation
    el10TgTransgenic Insertion
      el375TgTransgenic Insertion
        el520
          		Small Deletion
          el538
            		Indel
            el574
              		Small Deletion
              el586
                		Small Deletion
                el593
                  		Small Deletion
                  el595
                    		Small Deletion
                    el624
                      		Small Deletion
                      1 - 10 of 21
                      Show
                      Human Disease / Model
                      No data available
                      Sequence Targeting Reagents
                      Target Reagent Reagent Type
                      cd248aCRISPR1-cd248aCRISPR
                      cdh11TALEN1-cdh11TALEN
                      ccn2aCRISPR7-ccn2aCRISPR
                      ccn2bCRISPR1-ccn2bCRISPR
                      emx2TALEN1-emx2TALEN
                      fstaCRISPR1-fstaCRISPR
                      fstbCRISPR1-fstbCRISPR
                      her6TALEN1-her6TALEN
                      mrrfCRISPR1-mrrfCRISPR
                      osr1TALEN1-osr1TALEN
                      1 - 10 of 13
                      Show
                      Fish
                      Fish
                      ccn2ael654/el654
                      ccn2bel689/el689
                      cd248ael679/el679
                      cdh11el520/el520
                      dlx5aj1073aGt; el10Tg
                      edn1tf216b/tf216b
                      edn1tf216b/tf216b; el10Tg; y1Tg
                      edn1tf216b/tf216b; emx2el586/el586
                      el10Tg; pd24Tg
                      el10Tg; y1Tg
                      1 - 10 of 25
                      Show
                      Antibodies
                      No data available
                      Orthology
                      No data available
                      Engineered Foreign Genes
                      Marker Marker Type Name
                      DsRedEFGDsRed
                      EGFPEFGEGFP
                      1 - 2 of 2
                      Show
                      Mapping
                      No data available
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                      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.