PUBLICATION
Aberrant expression of genes necessary for neuronal development and notch signaling in an epileptic mind bomb zebrafish
- Authors
- Hortopan, G.A., and Baraban, S.C.
- ID
- ZDB-PUB-110629-14
- Date
- 2011
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 240(8): 1964-76 (Journal)
- Registered Authors
- Baraban, Scott
- Keywords
- brain, development, GFP, notch, zebrafish
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Embryo, Nonmammalian/anatomy & histology
- Embryo, Nonmammalian/physiology
- Epilepsy/physiopathology*
- Gene Expression Profiling
- Gene Expression Regulation, Developmental*
- Homeodomain Proteins/genetics
- Homeodomain Proteins/metabolism
- Microarray Analysis
- Neurogenesis/physiology
- Neurons/cytology
- Neurons/physiology*
- Receptors, Notch/genetics
- Receptors, Notch/metabolism*
- Signal Transduction/genetics*
- Ubiquitin-Protein Ligases/genetics
- Ubiquitin-Protein Ligases/metabolism*
- Zebrafish/anatomy & histology
- Zebrafish/embryology
- Zebrafish/physiology*
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 21688347 Full text @ Dev. Dyn.
Citation
Hortopan, G.A., and Baraban, S.C. (2011) Aberrant expression of genes necessary for neuronal development and notch signaling in an epileptic mind bomb zebrafish. Developmental Dynamics : an official publication of the American Association of Anatomists. 240(8):1964-76.
Abstract
Mutation within an ubiquitin E3 ligase gene can lead to a failure in Notch signaling, excessive neurons, and depletion of neural progenitor cells in mind bomb mutants. Using mib(hi904) zebrafish, we reported seizures and a down-regulation of γ-aminobutyric acid (GABA) signaling pathway genes. A transcriptome analysis also identified differential expression pattern of genes related to Notch signaling and neurodevelopment. Here, we selected nine of these genes (her4.2, hes5, bhlhb5, hoxa5a, hoxb5b, dmbx1a, dbx1a, nxph1, and plxnd1) and performed a more thorough analysis of expression using conventional polymerase chain reaction, real-time polymerase chain reaction and in situ hybridization. Transgenic reporter fish (Gfap:GFP and Dlx5a-6a:GFP) were used to assess early brain morphology in vivo. Down-regulation of many of these genes was prominent throughout key structures of the developing mib(hi904) zebrafish brain including, but not limited to, the pallium, ventral thalamus, and optic tectum. Brain expression of Dlx5a-6a and Gfap was also reduced. In conclusion, these expression studies indicate a general down-regulation of Notch signaling genes necessary for proper brain development and suggest that these mutant fish could provide valuable insights into neurological conditions, such as Angelman syndrome, associated with ubiquitin E3 ligase mutation.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping