PUBLICATION
Neurotoxicity of cGMP in the vertebrate retina: from the initial research on rd mutant mice to zebrafish genetic approaches
- Authors
- Iribarne, M., Masai, I.
- ID
- ZDB-PUB-170817-3
- Date
- 2017
- Source
- Journal of neurogenetics 31(3): 88-101 (Review)
- Registered Authors
- Iribarne, Maria, Masai, Ichiro
- Keywords
- Aipl1, Pde6c, Zebrafish, genetic mutants, photoreceptor degeneration, retina
- MeSH Terms
-
- Animals
- Cyclic GMP*/genetics
- Cyclic GMP*/metabolism
- Cyclic GMP*/toxicity
- Cyclic Nucleotide Phosphodiesterases, Type 6/genetics
- Disease Models, Animal*
- Humans
- Mice
- Mice, Mutant Strains
- Mutation/genetics
- Photoreceptor Cells, Vertebrate/metabolism
- Retina/drug effects*
- Retinal Degeneration/genetics*
- Vision, Ocular/genetics
- Zebrafish
- PubMed
- 28812418 Full text @ J. Neurogenet.
Citation
Iribarne, M., Masai, I. (2017) Neurotoxicity of cGMP in the vertebrate retina: from the initial research on rd mutant mice to zebrafish genetic approaches. Journal of neurogenetics. 31(3):88-101.
Abstract
Zebrafish are an excellent animal model for research on vertebrate development and human diseases. Sophisticated genetic tools including large-scale mutagenesis methodology make zebrafish useful for studying neuronal degenerative diseases. Here, we review zebrafish models of inherited ophthalmic diseases, focusing on cGMP metabolism in photoreceptors. cGMP is the second messenger of phototransduction, and abnormal cGMP levels are associated with photoreceptor death. cGMP concentration represents a balance between cGMP phosphodiesterase 6 (PDE6) and guanylate cyclase (GC) activities in photoreceptors. Various zebrafish cGMP metabolism mutants were used to clarify molecular mechanisms by which dysfunctions in this pathway trigger photoreceptor degeneration. Here, we review the history of research on the retinal degeneration (rd) mutant mouse, which carries a genetic mutation of PDE6b, and we also highlight recent research in photoreceptor degeneration using zebrafish models. Several recent discoveries that provide insight into cGMP toxicity in photoreceptors are discussed.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping