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. T HE zebrafish has been an important model through apparent by the 18-19 SS. The first postmitotic neurons of the retina are generated at 28 hr postfertilization which genes necessary for visual system development and function have been identified (reviewed in (hpf) and by 72 hpf the retina is functional (Easter and Nicola 1996; Hu and Easter 1999; Schmitt and Easter and Malicki 2002 and Neuhauss 2003). Zebrafish eyes are large, easily accessible, and structurally Dowling 1999). Retinas of many fish and amphibians also possess a specialized region at their margins, termed similar to the human eye. Eye formation in zebrafish is analogous to that observed in other vertebrate embryos, peripheral or ciliary marginal zones, that perpetually adds cells to the retina during the lifetime of the animal thus providing an excellent model system with which the understanding of vertebrate eye development can ( Johns 1977). Several generations of chemically based forward gebe advanced. Additionally, many disrupted genes and pathways identified as integral to the formation of the netic screens have been undertaken in zebrafish (Driever et al. 1996;Haffter et al. 1996; Matsuda and Mishina zebrafish eye produce phenotypes that resemble disorders of the human visual system. Thus, characterization 2004), some of which have focused on eye development and function (Malicki et al. 1996; Fadool et al. 1997; of the molecular mechanisms of eye development in zebrafish should facilitate a better understanding of these hu- Neuhauss et al. 1999). While these chemically based screens have been instrumental in generating interesting man pathologies (Goldsmith and Harris 2003).Eye development in zebrafish ...