PUBLICATION

Zebrafish zic2 controls formation of periocular neural crest and choroid fissure morphogenesis

Authors
Sedykh, I., Yoon, B., Roberson, L., Moskvin, O., Dewey, C.N., Grinblat, Y.
ID
ZDB-PUB-170712-18
Date
2017
Source
Developmental Biology   429(1): 92-104 (Journal)
Registered Authors
Grinblat, Yevgenya, Roberson, Laura
Keywords
Hedgehog signaling, alx1, coloboma, zebrafish, zic2
Datasets
GEO:GSE99382
MeSH Terms
  • Cell Lineage/drug effects
  • Cell Lineage/genetics
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Mutation/genetics
  • Sequence Analysis, RNA
  • Gene Expression Regulation, Developmental/drug effects
  • PAX2 Transcription Factor/genetics
  • PAX2 Transcription Factor/metabolism
  • Animals
  • Neural Crest/cytology
  • Neural Crest/drug effects
  • Neural Crest/metabolism*
  • Cartilage/drug effects
  • Cartilage/metabolism
  • Skull/embryology
  • Veratrum Alkaloids/pharmacology
  • Coloboma/pathology
  • Face/embryology
  • Gene Expression Profiling
  • Zebrafish/embryology
  • Zebrafish/genetics
  • Zebrafish/metabolism*
  • Choroid/embryology*
  • Choroid/metabolism*
  • Morphogenesis*/drug effects
  • Morphogenesis*/genetics
  • Transcription Factors/genetics
  • Transcription Factors/metabolism*
  • Sequence Homology, Amino Acid
  • Retina/drug effects
  • Retina/embryology
(all 32)
PubMed
28689736 Full text @ Dev. Biol.
Abstract
The vertebrate retina develops in close proximity to the forebrain and neural crest-derived cartilages of the face and jaw. Coloboma, a congenital eye malformation, is associated with aberrant forebrain development (holoprosencephaly) and with craniofacial defects (frontonasal dysplasia) in humans, suggesting a critical role for cross-lineage interactions during retinal morphogenesis. ZIC2, a zinc-finger transcription factor, is linked to human holoprosencephaly. We have previously used morpholino assays to show zebrafish zic2 functions in the developing forebrain, retina and craniofacial cartilage. We now report that zebrafish with genetic lesions in zebrafish zic2 orthologs, zic2a and zic2b, develop with retinal coloboma and craniofacial anomalies. We demonstrate a requirement for zic2 in restricting pax2a expression and show evidence that zic2 function limits Hh signaling. RNA-seq transcriptome analysis identified an early requirement for zic2 in periocular neural crest as an activator of alx1, a transcription factor with essential roles in craniofacial and ocular morphogenesis in human and zebrafish. Collectively, these data establish zic2 mutant zebrafish as a powerful new genetic model for in-depth dissection of cell interactions and genetic controls during craniofacial complex development.
Genes / Markers
Figures
Figure Gallery (10 images)
Show all Figures
Expression
No data available
Phenotype
Fish Conditions Stage Phenotype Figure
zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gtstandard conditionsDay 4
zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gtstandard conditionsDay 4
zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gtstandard conditionsDay 5
zic2buwt104/uwt104; zic2amn0133Gt/mn0133Gtstandard conditionsDay 4
zic2buwt104/uwt104; zic2amn0133Gt/mn0133Gtstandard conditionsDay 4
1 - 5 of 5
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
mn0133GtTransgenic Insertion
uw1116
    		Insertion
    uw1127
      		Insertion
      uwt104
        		Small Deletion
        1 - 4 of 4
        Show
        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        zic2bCRISPR1-zic2bCRISPR
        zic2bTALEN1-zic2bTALEN
        1 - 2 of 2
        Show
        Fish
        Fish
        zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gt
        zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gt
        zic2buw1127/uw1127; zic2amn0133Gt/mn0133Gt
        zic2buwt104/uwt104; zic2amn0133Gt/mn0133Gt
        1 - 4 of 4
        Show
        Antibodies
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        GFPEFGGFP
        mRFP1EFGmRFP1
        1 - 2 of 2
        Show
        Mapping
        No data available
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        Citation
        Sedykh, I., Yoon, B., Roberson, L., Moskvin, O., Dewey, C.N., Grinblat, Y. (2017) Zebrafish zic2 controls formation of periocular neural crest and choroid fissure morphogenesis. Developmental Biology. 429(1):92-104.