PUBLICATION
The occhiolino (occ) mutant zebrafish, a model for development of the optical function in the biological lens
- Authors
- Aose, M., Linbo, T.H., Lawrence, O., Senoo, T., Raible, D.W., Clark, J.I.
- ID
- ZDB-PUB-170420-6
- Date
- 2017
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 246(11): 915-924 (Journal)
- Registered Authors
- Raible, David
- Keywords
- none
- MeSH Terms
-
- Animals
- Disease Models, Animal
- Electroretinography
- Embryo, Nonmammalian
- Eye Abnormalities/genetics
- Immunohistochemistry
- Laminin
- Lens Capsule, Crystalline/anatomy & histology
- Lens Capsule, Crystalline/pathology
- Lens, Crystalline/embryology
- Lens, Crystalline/growth & development*
- Zebrafish/embryology
- Zebrafish/genetics
- PubMed
- 28422363 Full text @ Dev. Dyn.
Citation
Aose, M., Linbo, T.H., Lawrence, O., Senoo, T., Raible, D.W., Clark, J.I. (2017) The occhiolino (occ) mutant zebrafish, a model for development of the optical function in the biological lens. Developmental Dynamics : an official publication of the American Association of Anatomists. 246(11):915-924.
Abstract
Background Zebrafish visual function depends on quality optics. An F3 screen for developmental mutations in the zebrafish nervous system was conducted in wild-type (wt) AB zebrafish exposed to 3 mM of N-ethyl-N-nitrosourea (ENU).
Results Mutant offspring, identified in an F3 screen, were characterized by a small pupil, resulting from retinal hypertrophy or hyperplasia and a small lens. Deficits in visual function made feeding difficult after hatching at approximately 5-6 dpf (days post fertilization). Special feeding conditions were necessary for survival of the occhiolino (occ) mutants after 6dpf. Optokinetic (OKR) tests measured defects in visual function in the occ mutant, although electroretinograms (ERG) were normal in the mutant and wt. Consistent with the ERGs, histology found normal retinal structure in the occ mutant and wt zebrafish. However, lens development was abnormal. Multiphoton imaging of the developmental stages of live embryos confirmed the formation of a secondary mass of lens cells in the developing eye of the mutant zebra fish at 3-4 dpf and laminin immunohistochemistry indicated the lens capsule was thin and disorganized in the mutant zebrafish.
Conclusion The occ zebrafish is a novel disease model for visual defects associated with abnormal lens development. This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping