A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon
Heisenberg C-PJ, Houart C, Take Uchi M, Rauch G, Young N, Coutinho P, Masai I, Caneparo L, Concha M, Geisler R, Dale T, Wilson S, Stemple D. 2001. A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon. Genes and Development. 15(11), 1427–1434.
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Author
Heisenberg, Carl-Philipp ISTA 
;
Houart, Corinne;
Take Uchi, Masaya;
Rauch, Gerd;
Young, Neville;
Coutinho, Pedro;
Masai, Ichiro;
Caneparo, Luca;
Concha, Miguel;
Geisler, Robert;
Dale, Trevor;
Wilson, Stephen
All
;
Houart, Corinne;
Take Uchi, Masaya;
Rauch, Gerd;
Young, Neville;
Coutinho, Pedro;
Masai, Ichiro;
Caneparo, Luca;
Concha, Miguel;
Geisler, Robert;
Dale, Trevor;
Wilson, Stephen
All
Abstract
Zebrafish embryos homozygous for the masterblind (mb1) 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 mb1(-/-) 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 Gsk3 beta can restore eye and telencephalic fates to mb1(-/-) 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.
Publishing Year
Date Published
2001-06-01
Journal Title
Genes and Development
Publisher
Cold Spring Harbor Laboratory Press
Acknowledgement
We thank many colleagues who provided reagents that enabled us to test axin1 and several other genes as candidates for the mbl mutation. In particular, we are indebted to Masahiko Hibi, Ken Irvine, Antonio Jacinto, Yun-Jin Jiang, Julian Lewis, and Tom Vogt for help and advice. We thank Ajay Chitnis and Dana Zivkovic for providing information prior to publication. This study was supported primarily by grants from the EMBO and EC to C.P.H., Wellcome Trust and EC to S.W.W., from the MRC to D.S., from Naito to M.T., from the DHGP to G.J.R. and R.G., and from the CRC/ICR to T.D. P.C. was supported by a PhD studentship from Fundação para a Ciência e Tecnologia, Programa PRAXIS XXI. S.W.W. is a Wellcome Trust Senior Research Fellow.
The publication costs of this article were defrayed in part by payment of page charges. This article must therefore be hereby marked “advertisement” in accordance with 18 USC section 1734 solely to indicate this fact.
Volume
15
Issue
11
Page
1427 - 1434
ISSN
IST-REx-ID
Cite this
Heisenberg C-PJ, Houart C, Take Uchi M, et al. A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon. Genes and Development. 2001;15(11):1427-1434. doi:10.1101/gad.194301
Heisenberg, C.-P. J., Houart, C., Take Uchi, M., Rauch, G., Young, N., Coutinho, P., … Stemple, D. (2001). A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon. Genes and Development. Cold Spring Harbor Laboratory Press. https://doi.org/10.1101/gad.194301
Heisenberg, Carl-Philipp J, Corinne Houart, Masaya Take Uchi, Gerd Rauch, Neville Young, Pedro Coutinho, Ichiro Masai, et al. “A Mutation in the Gsk3-Binding Domain of Zebrafish Masterblind/Axin1 Leads to a Fate Transformation of Telencephalon and Eyes to Diencephalon.” Genes and Development. Cold Spring Harbor Laboratory Press, 2001. https://doi.org/10.1101/gad.194301.
C.-P. J. Heisenberg et al., “A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon,” Genes and Development, vol. 15, no. 11. Cold Spring Harbor Laboratory Press, pp. 1427–1434, 2001.
Heisenberg C-PJ, Houart C, Take Uchi M, Rauch G, Young N, Coutinho P, Masai I, Caneparo L, Concha M, Geisler R, Dale T, Wilson S, Stemple D. 2001. A mutation in the Gsk3-binding domain of zebrafish Masterblind/Axin1 leads to a fate transformation of telencephalon and eyes to diencephalon. Genes and Development. 15(11), 1427–1434.
Heisenberg, Carl-Philipp J., et al. “A Mutation in the Gsk3-Binding Domain of Zebrafish Masterblind/Axin1 Leads to a Fate Transformation of Telencephalon and Eyes to Diencephalon.” Genes and Development, vol. 15, no. 11, Cold Spring Harbor Laboratory Press, 2001, pp. 1427–34, doi:10.1101/gad.194301.
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