PUBLICATION
A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon
- Authors
- Heisenberg, C.P., Houart, C., Take-uchi, M., Rauch, G.J., Young, N., Coutinho, P., Masai, I., Caneparo, L., Concha, M.L., Geisler, R., Dale, T.C., Wilson, S.W., and Stemple, D.L.
- ID
- ZDB-PUB-010705-2
- Date
- 2001
- Source
- Genes & Development 15(11): 1427-1434 (Journal)
- Registered Authors
- Caneparo, Luca, Concha, Miguel, Coutinho, Pedro, Geisler, Robert, Heisenberg, Carl-Philipp, Houart, Corinne, Masai, Ichiro, Rauch, Gerd-Jörg, Stemple, Derek L., Take-uchi, Masaya, Wilson, Steve
- Keywords
- Axin; Wnt; forebrain; zebrafish
- MeSH Terms
-
- Zebrafish
- Diencephalon/embryology*
- Diencephalon/growth & development
- Diencephalon/metabolism
- Eye/embryology*
- Eye/metabolism
- Protein Binding
- Zebrafish Proteins*
- Repressor Proteins*
- Axin Protein
- Precipitin Tests
- Mutation
- Signal Transduction
- Conserved Sequence
- Body Patterning/genetics*
- Body Patterning/physiology
- Telencephalon/embryology*
- Telencephalon/growth & development
- Telencephalon/metabolism
- Animals
- In Situ Hybridization
- Embryo, Nonmammalian
- Proteins/genetics*
- Proteins/metabolism
- Protein Structure, Tertiary
- Glycogen Synthase Kinase 3
- Proto-Oncogene Proteins/genetics
- Proto-Oncogene Proteins/physiology
- Wnt Proteins
- Calcium-Calmodulin-Dependent Protein Kinases/genetics*
- Calcium-Calmodulin-Dependent Protein Kinases/metabolism
- PubMed
- 11390362 Full text @ Genes & Dev.
Citation
Heisenberg, C.P., Houart, C., Take-uchi, M., Rauch, G.J., Young, N., Coutinho, P., Masai, I., Caneparo, L., Concha, M.L., Geisler, R., Dale, T.C., Wilson, S.W., and Stemple, D.L. (2001) A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon. Genes & Development. 15(11):1427-1434.
Abstract
Zebrafish embryos homozygous for the masterblind (mbl) mutation exhibit a striking phenotype in which the eyes and telencephalon are reduced or absent and diencephalic fates expand to the front of the brain. Here we show that mbl(-/-) embryos carry an amino-acid change at a conserved site in the Wnt pathway scaffolding protein, Axin1. The amino-acid substitution present in the mbl allele abolishes the binding of Axin to Gsk3 and affects Tcf-dependent transcription. Therefore, Gsk3 activity may be decreased in mbl(-/-) embryos and in support of this possibility, overexpression of either wild-type Axin1 or Gsk3beta can restore eye and telencephalic fates to mbl(-/-) embryos. Our data reveal a crucial role for Axin1-dependent inhibition of the Wnt pathway in the early regional subdivision of the anterior neural plate into telencephalic, diencephalic, and eye-forming territories.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping