PUBLICATION
Comparative synteny cloning of zebrafish you-too: mutations in the Hedgehog target gli2 affect ventral forebrain patterning
- Authors
- Karlstrom, R.O., Talbot, W.S., and Schier, A.F.
- ID
- ZDB-PUB-990302-44
- Date
- 1999
- Source
- Genes & Development 13: 388-393 (Journal)
- Registered Authors
- Karlstrom, Rolf, Schier, Alexander, Talbot, William S.
- Keywords
- zebrafish mutations; Hh signaling; gli2; ventral forebrain
- MeSH Terms
-
- In Situ Hybridization
- Embryonic Development
- Molecular Sequence Data
- Zebrafish/embryology*
- Signal Transduction/genetics
- PubMed
- 10049354 Full text @ Genes & Dev.
Abstract
Zebrafish you-too (yot) mutations interfere with Hedgehog (Hh) signaling during embryogenesis. Using a comparative synteny approach, we isolated yot as a zinc finger transcription factor homologous to the Hh target gli2. Two alleles of yot contain nonsense mutations resulting in carboxy-terminally truncated proteins. In addition to causing defects in midline development, muscle differentiation, and retinal axon guidance, yot mutations disrupt anterior pituitary and ventral forebrain differentiation. yot mutations also cause ectopic lens formation in the ventral diencephalon. These findings reveal that truncated zebrafish Gli2 proteins interfere with Hh signaling necessary for differentiation and axon guidance in the ventral forebrain.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping