PUBLICATION
Retinal defects in the zebrafish bleached mutant
- Authors
- Neuhauss, S.C., Seeliger, M.W., Schepp, C.P., and Biehlmaier, O.
- ID
- ZDB-PUB-030812-5
- Date
- 2003
- Source
- Documenta ophthalmologica. Advances in ophthalmology 107(1): 71-78 (Journal)
- Registered Authors
- Biehlmaier, Oliver, Neuhauss, Stephan
- Keywords
- none
- MeSH Terms
-
- Animals
- Apoptosis
- Blindness/etiology
- Electroretinography
- Eye Proteins/genetics
- Genes, Recessive
- Larva
- Mutation*
- Retina/growth & development
- Retina/pathology
- Retina/physiopathology
- Retinal Degeneration/genetics*
- Retinal Degeneration/pathology
- Retinal Degeneration/physiopathology
- Zebrafish/genetics*
- Zebrafish/growth & development
- Zebrafish Proteins/genetics*
- PubMed
- 12906124 Full text @ Doc. Ophthalmol.
Citation
Neuhauss, S.C., Seeliger, M.W., Schepp, C.P., and Biehlmaier, O. (2003) Retinal defects in the zebrafish bleached mutant. Documenta ophthalmologica. Advances in ophthalmology. 107(1):71-78.
Abstract
The recessive zebrafish mutant bleached has, apart from its defects in pigmentation, a heritable defect leading to larval blindness. Here, we analyze the retina of homozygous bleached larvae, employing morphological and electrophysiological methods. Electroretinography revealed a complete lack of electrical signals in response to light. Histological analysis of mutant retinae showed a severely affected outer retina with a hypopigmented pigment epithelium and a disorganized outer nuclear layer containing few or no intact photoreceptors. Using the TUNEL assay for cell death detection, we noticed a strong increase of apoptotic cells in all retinal cell layers, starting in young larvae even before retinal support of visual function. At later stages cell death is most pronounced at the marginal zone, where new cells are constantly added to the retina. At early stages increased apoptosis is mainly confined to the retina, while at later stages elevated cell death is al so apparent in extra-retinal tissues, particularly in the brain. Hence, the lack of visual responses in homozygous bleached larvae can be attributed to a severe defect of the outer retina, preceded by increased levels of apoptotic cell death in all retinal cell layers.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping