PUBLICATION
Zebrafish Models of Photoreceptor Dysfunction and Degeneration
- Authors
- Noel, N.C.L., MacDonald, I.M., Allison, W.T.
- ID
- ZDB-PUB-210114-2
- Date
- 2021
- Source
- Biomolecules 11(1): (Review)
- Registered Authors
- Allison, Ted
- Keywords
- Danio rerio, Leber congenital amaurosis, choroideremia, cone dystrophy, cone-rod dystrophy, inherited photoreceptor disease, macular degeneration, regeneration, retinal neovascularization, retinitis pigmentosa
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Disease Models, Animal
- Models, Biological
- Mutation/genetics
- Photoreceptor Cells, Vertebrate/pathology*
- Retinal Degeneration/genetics
- Retinal Degeneration/pathology*
- Retinal Degeneration/physiopathology*
- Retinal Degeneration/therapy
- Zebrafish
- PubMed
- 33435268 Full text @ Biomolecules
Citation
Noel, N.C.L., MacDonald, I.M., Allison, W.T. (2021) Zebrafish Models of Photoreceptor Dysfunction and Degeneration. Biomolecules. 11(1):.
Abstract
Zebrafish are an instrumental system for the generation of photoreceptor degeneration models, which can be utilized to determine underlying causes of photoreceptor dysfunction and death, and for the analysis of potential therapeutic compounds, as well as the characterization of regenerative responses. We review the wealth of information from existing zebrafish models of photoreceptor disease, specifically as they relate to currently accepted taxonomic classes of human rod and cone disease. We also highlight that rich, detailed information can be derived from studying photoreceptor development, structure, and function, including behavioural assessments and in vivo imaging of zebrafish. Zebrafish models are available for a diversity of photoreceptor diseases, including cone dystrophies, which are challenging to recapitulate in nocturnal mammalian systems. Newly discovered models of photoreceptor disease and drusenoid deposit formation may not only provide important insights into pathogenesis of disease, but also potential therapeutic approaches. Zebrafish have already shown their use in providing pre-clinical data prior to testing genetic therapies in clinical trials, such as antisense oligonucleotide therapy for Usher syndrome.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping