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Originally published as Genetics Published Articles Ahead of Print on February 16, 2005.
Genetics, Vol. 170, 245-261, May 2005, Copyright © 2005
doi:10.1534/genetics.104.039727
Identification of Zebrafish Insertional Mutants With Defects in Visual System Development and Function
Jeffrey M. Gross*,1,
Brian D. Perkins*,2,
Adam Amsterdam
,
Ana Egaña*,
Tristan Darland*,
Jonathan I. Matsui*,
Salvatore Sciascia*,
Nancy Hopkins and
John E. Dowling*
* Department of Molecular and Cellular Biology, Harvard University, Cambridge, Massachusetts 02138
Center for Cancer Research and Department of Biology, Massachusetts Institute of Technology, Cambridge, Massachusetts 02139
1 Corresponding author: Department of Molecular and Cellular Biology, Harvard University, 16 Divinity Ave., Cambridge, MA 02138.
E-mail: jmgross{at}fas.harvard.edu
Genetic analysis in zebrafish has been instrumental in identifying genes necessary for visual system development and function. Recently, a large-scale retroviral insertional mutagenesis screen, in which 315 different genes were mutated, that resulted in obvious phenotypic defects by 5 days postfertilization was completed. That the disrupted gene has been identified in each of these mutants provides unique resource through which the formation, function, or physiology of individual organ systems can be studied. To that end, a screen for visual system mutants was performed on 250 of the mutants in this collection, examining each of them histologically for morphological defects in the eye and behaviorally for overall visual system function. Forty loci whose disruption resulted in defects in eye development and/or visual function were identified. The mutants have been divided into the following phenotypic classes that show defects in: (1) morphogenesis, (2) growth and central retinal development, (3) the peripheral marginal zone, (4) retinal lamination, (5) the photoreceptor cell layer, (6) the retinal pigment epithelium, (7) the lens, (8) retinal containment, and (9) behavior. The affected genes in these mutants highlight a diverse set of proteins necessary for the development, maintenance, and function of the vertebrate visual system.
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