PUBLICATION

Zebrafish churchill regulates developmental gene expression and cell migration

Authors
Taibi, A., Mandavawala, K.P., Noel, J., Okoye, E.V., Milano, C.R., Martin, B.L., and Sirotkin, H.I.
ID
ZDB-PUB-130312-20
Date
2013
Source
Developmental Dynamics : an official publication of the American Association of Anatomists   242(6): 614-21 (Journal)
Registered Authors
Martin, Benjamin, Sirotkin, Howard
Keywords
churchill, zinc finger nuclease, cell migration, zebrafish
MeSH Terms
  • Alleles
  • Animals
  • Body Patterning
  • Cell Movement
  • Fibroblast Growth Factors/metabolism
  • Gene Expression Regulation, Developmental*
  • In Situ Hybridization
  • Mutation
  • Nodal Protein/metabolism
  • Signal Transduction
  • Trans-Activators/genetics
  • Trans-Activators/metabolism*
  • Trans-Activators/physiology*
  • Transforming Growth Factor beta/metabolism
  • Transgenes
  • Xenopus Proteins/genetics
  • Xenopus Proteins/physiology*
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Zinc Fingers
PubMed
23443939 Full text @ Dev. Dyn.
Abstract

Background: Regulation of developmental signaling pathways is essential for embryogenesis. The small putative zinc finger protein, Churchill (ChCh) has been implicated in modulation of both TGF-β and FGF signaling.

Results: We employed zinc finger nuclease (ZFN) mediated gene targeting to disrupt the zebrafish chch locus and generate the first chch mutations. Three induced lesions produce frameshift mutations that truncate the protein in the third of five β-strands that comprise the protein. Surprisingly, zygotic and maternal zygotic chch mutants are viable. Mutants have elevated expression of mesodermal markers but progress normally through early development. chch mutants are sensitive to exogenous Nodal. However, neither misregulation of FGF targets nor sensitivity to exogenous FGF was detected. Finally, chch mutant cells were found to undergo inappropriate migration in cell transplant assays.

Conclusions: Together, these results suggest that chch is not essential for survival, but functions to modulate early mesendodermal gene expression and limit cell migration.

Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping