PUBLICATION
The Bugeye Mutant Zebrafish Exhibits Visual Deficits that Arise with the Onset of an Enlarged Eye Phenotype
- Authors
- Stujenske, J.M., Dowling, J.E., and Emran, F.
- ID
- ZDB-PUB-110503-7
- Date
- 2011
- Source
- Investigative ophthalmology & visual science 52(7): 4200-4207 (Journal)
- Registered Authors
- Dowling, John E., Emran, Farida
- Keywords
- none
- MeSH Terms
-
- Aging
- Animals
- Contrast Sensitivity
- Disease Models, Animal*
- Electroretinography
- Eye/pathology
- Eye Abnormalities/diagnosis
- Eye Abnormalities/genetics*
- Eye Abnormalities/pathology*
- Eye Abnormalities/physiopathology
- Hypertrophy
- Intraocular Pressure
- Larva
- Mutation*
- Oculomotor Nerve/physiopathology
- Phenotype
- Photic Stimulation/methods
- Reaction Time
- Retina/pathology
- Retina/physiopathology
- Retinal Ganglion Cells
- Vision Disorders/genetics*
- Zebrafish/genetics*
- Zebrafish/growth & development
- PubMed
- 21460263 Full text @ Invest. Ophthalmol. Vis. Sci.
Citation
Stujenske, J.M., Dowling, J.E., and Emran, F. (2011) The Bugeye Mutant Zebrafish Exhibits Visual Deficits that Arise with the Onset of an Enlarged Eye Phenotype. Investigative ophthalmology & visual science. 52(7):4200-4207.
Abstract
Purpose: The bugeye mutant has an enlarged eye phenotype presumably because of elevated intraocular pressure. Since elevated intraocular pressure is a significant risk factor for glaucoma, the bugeye zebrafish mutant may be a model organism for the disease. Methods: The optomotor response (OMR) was used to assess visual responsiveness in both larval (1) and adult zebrafish (2). Electroretinograms (ERGs) were recorded to measure outer retinal function (3) and histological analyses were performed on WT and mutant eyes(4). Results: At 5 days old, bugeye mutants have an OMR, ERGs, and retinal morphology indistinguishable from wildtype (WT) animals. By 2 months of age, bugeye mutants begin to develop an enlarged eye phenotype. At 3 months, some mutants show deficits in the OMR assay, including lower contrast sensitivity. Our data suggest that there is a correlation between the size of the enlarged eye and the degree of OMR deficit. Histological analysis of the bugeye mutant retina reveals decreases in retinal ganglion cell densities by 3 months. By 5 months, the ERG b-wave has smaller amplitudes and longer latencies at brighter light intensities as compared with those of WT fish. Conclusions: Following phenotypic onset at 3 months, the bugeye mutants begin to develop visual deficits. At 3 months, bugeye mutants exhibit a decrease in retinal cell densities and by 5 months, show diminished outer retinal function. In summary, the bugeye mutant provides a means for studying glaucoma-associated phenotypes in the zebrafish.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping