| ZFIN ID: ZDB-PUB-010705-2 |
A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon
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.
| 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: |
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| 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.
ADDITIONAL INFORMATION
- Genes / Markers (7)
- Mutations and Transgenics (1)
- Fish (2)
- Orthology (2)