PUBLICATION
An expanded domain of fgf3 expression in the hindbrain of zebrafish valentino mutants results in mis-patterning of the otic vesicle
- Authors
- Kwak, S.-J., Phillips, B.T., Heck R., and Riley, B.B.
- ID
- ZDB-PUB-021105-12
- Date
- 2002
- Source
- Development (Cambridge, England) 129(22): 5279-5287 (Journal)
- Registered Authors
- Kwak, Su-Jin, Riley, Bruce
- Keywords
- inner ear; hair cell; FGF signaling; ace; morpholino; val; kreisler; pax5; zebrafish
- MeSH Terms
-
- Zebrafish/embryology*
- Zebrafish/genetics*
- PAX5 Transcription Factor
- Animals
- Fibroblast Growth Factor 8
- Transcription Factors/genetics
- Transcription Factors/metabolism
- Fibroblast Growth Factors/genetics
- Fibroblast Growth Factors/metabolism*
- Hair/abnormalities
- Homeodomain Proteins/genetics
- Homeodomain Proteins/metabolism
- Embryo, Nonmammalian
- Mutation
- Nerve Tissue Proteins/genetics
- Nerve Tissue Proteins/metabolism
- Proto-Oncogene Proteins/genetics
- Proto-Oncogene Proteins/metabolism*
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- Gene Expression Regulation, Developmental
- Fibroblast Growth Factor 3
- DNA-Binding Proteins/genetics
- DNA-Binding Proteins/metabolism
- Rhombencephalon/embryology*
- Rhombencephalon/metabolism
- Body Patterning/genetics
- Ear, Inner/abnormalities
- Ear, Inner/embryology*
- PubMed
- 12399318 Full text @ Development
Citation
Kwak, S.-J., Phillips, B.T., Heck R., and Riley, B.B. (2002) An expanded domain of fgf3 expression in the hindbrain of zebrafish valentino mutants results in mis-patterning of the otic vesicle. Development (Cambridge, England). 129(22):5279-5287.
Abstract
The valentino (val) mutation in zebrafish perturbs hindbrain patterning and, as a secondary consequence, also alters development of the inner ear. We have examined the relationship between these defects and expression of fgf3 and fgf8 in the hindbrain. The otic vesicle in val/ val mutants is smaller than normal, yet produces nearly twice the normal number of hair cells, and some hair cells are produced ectopically between the anterior and posterior maculae. Anterior markers pax5 and nkx5.1 are expressed in expanded domains that include the entire otic epithelium juxtaposed to the hindbrain, and the posterior marker zp23 is not expressed. In the mutant hindbrain, expression of fgf8 is normal, whereas the domain of fgf3 expression expands to include rhombomere 4 through rhombomere X (an aberrant segment that forms in lieu of rhombomeres 5 and 6). Depletion of fgf3 by injection of antisense morpholino (fgf3-MO) suppresses the ear patterning defects in val/val embryos: Excess and ectopic hair cells are eliminated, expression of anterior otic markers is reduced or ablated, and zp23 is expressed throughout the medial wall of the otic vesicle. By contrast, disruption of fgf8 does not suppress the val/val phenotype but instead interacts additively, indicating that these genes affect distinct developmental pathways. Thus, the inner ear defects observed in val/val mutants appear to result from ectopic expression of fgf3 in the hindbrain. These data also indicate that val normally represses fgf3 expression in r5 and r6, an interpretation further supported by the effects of misexpressing val in wild-type embryos. This is in sharp contrast to the mouse, in which fgf3 is normally expressed in r5 and r6 because of positive regulation by kreisler, the mouse ortholog of val. Implications for co-evolution of the hindbrain and inner ear are discussed.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping