title: Identification of zebrafish insertional mutants with defects in visual system development and function.
abstract: 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.
authors.affiliation: Department of Molecular and Cellular Biology, Harvard University, Cambridge, Massachusetts 02138, USA. jmgross@fas.harvard.edu
authors.forename: Jeffrey M
authors.initials: JM
authors.lastname: Gross
authors.forename: Brian D
authors.initials: BD
authors.lastname: Perkins
authors.forename: Adam
authors.initials: A
authors.lastname: Amsterdam
authors.forename: Ana
authors.initials: A
authors.lastname: Egaña
authors.forename: Tristan
authors.initials: T
authors.lastname: Darland
authors.forename: Jonathan I
authors.initials: JI
authors.lastname: Matsui
authors.forename: Salvatore
authors.initials: S
authors.lastname: Sciascia
authors.forename: Nancy
authors.initials: N
authors.lastname: Hopkins
authors.forename: John E
authors.initials: JE
authors.lastname: Dowling
mesh_terms: D000818:Animals; D001522:Behavior, Animal; D002470:Cell Survival / Q000235:genetics; D005123:Eye / Q000033:anatomy & histology / Q000196:embryology* / Q000254:growth & development; D005124:Eye Abnormalities / Q000235:genetics* / Q000378:metabolism; D007908:Lens, Crystalline / Q000002:abnormalities; D009154:Mutation; D009799:Ocular Physiological Phenomena; D010786:Photoreceptor Cells / Q000473:pathology; D012160:Retina / Q000473:pathology; D015027:Zebrafish / Q000033:anatomy & histology / Q000235:genetics* / Q000254:growth & development / Q000502:physiology*
references.citation: Cell Prolif. 1995 Dec;28(12):645-57
references.pmid: 8634372
references.citation: Results Probl Cell Differ. 2002;40:346-70
references.pmid: 12353485
references.citation: Nature. 1996 Oct 31;383(6603):829-32
references.pmid: 8893009
references.citation: Dev Biol. 1996 Dec 15;180(2):646-63
references.pmid: 8954734
references.citation: Genes Dev. 1996 Dec 15;10(24):3141-55
references.pmid: 8985183
references.citation: Development. 1996 Dec;123:1-36
references.pmid: 9007226
references.citation: Proc Natl Acad Sci U S A. 2002 Nov 12;99(23):14682-7
references.pmid: 12391305
references.citation: J Neurobiol. 2003 Jan;54(1):148-60
references.pmid: 12486702
references.citation: Proc Natl Acad Sci U S A. 2003 Nov 25;100(24):14109-14
references.pmid: 14610273
references.citation: Proc Natl Acad Sci U S A. 2004 Feb 3;101(5):1374-9
references.pmid: 14734810
references.citation: FASEB J. 2004 Mar;18(3):562-4
references.pmid: 14715704
references.citation: Development. 2004 May;131(9):1913-25
references.pmid: 15056614
references.citation: Dev Cell. 2004 Jun;6(6):781-90
references.pmid: 15177027
references.citation: Neuron. 2004 Jun 10;42(5):703-16
references.pmid: 15182712
references.citation: Development. 2004 Aug;131(16):4085-93
references.pmid: 15269167
references.citation: Proc Natl Acad Sci U S A. 2004 Aug 31;101(35):12792-7
references.pmid: 15256591
references.citation: J Comp Neurol. 1977 Dec 1;176(3):343-57
references.pmid: 915042
references.citation: Am J Ophthalmol. 1980 Dec;90(6):783-91
references.pmid: 7446665
references.citation: Science. 1988 Mar 4;239(4844):1142-5
references.pmid: 2449732
references.citation: Nature. 1991 Mar 28;350(6316):339-41
references.pmid: 2008211
references.citation: EMBO J. 1994 Apr 1;13(7):1729-40
references.pmid: 8157011
references.citation: J Comp Neurol. 1994 Jun 22;344(4):532-42
references.pmid: 7929890
references.citation: Proc Natl Acad Sci U S A. 1995 Nov 7;92(23):10545-9
references.pmid: 7479837
references.citation: Dev Dyn. 1995 Jul;203(3):253-310
references.pmid: 8589427
references.citation: Trends Cell Biol. 2002 Dec;12(12):551-5
references.pmid: 12495842
references.citation: Semin Cell Dev Biol. 2003 Feb;14(1):11-8
references.pmid: 12524002
references.citation: Dev Dyn. 2003 Mar;226(3):570-7
references.pmid: 12619142
references.citation: Dev Biol. 2003 Jul 1;259(1):95-108
references.pmid: 12812791
references.citation: J Comp Neurol. 2003 Aug 25;463(3):265-80
references.pmid: 12820161
references.citation: Development. 1996 Dec;123:37-46
references.pmid: 9007227
references.citation: Development. 1996 Dec;123:191-203
references.pmid: 9007240
references.citation: Development. 1996 Dec;123:263-73
references.pmid: 9007246
references.citation: J Comp Neurol. 1997 May 19;381(4):449-60
references.pmid: 9136802
references.citation: Dev Genet. 1997;20(3):288-95
references.pmid: 9216068
references.citation: Trends Biochem Sci. 1998 May;23(5):174-8
references.pmid: 9612081
references.citation: Vision Res. 1998 May;38(10):1335-9
references.pmid: 9667001
references.citation: Proc Natl Acad Sci U S A. 1998 Aug 18;95(17):9932-7
references.pmid: 9707578
references.citation: Nature. 1998 Sep 10;395(6698):185-9
references.pmid: 9744278
references.citation: Development. 1998 Nov;125(22):4369-78
references.pmid: 9778497
references.citation: J Comp Neurol. 1999 Feb 22;404(4):515-36
references.pmid: 9987995
references.citation: Dev Biol. 1999 Mar 15;207(2):309-21
references.pmid: 10068465
references.citation: J Neurosci. 1999 Oct 1;19(19):8603-15
references.pmid: 10493760
references.citation: Dev Dyn. 2003 Nov;228(3):523-34
references.pmid: 14579390
references.citation: Genes Dev. 1999 Oct 15;13(20):2713-24
references.pmid: 10541557
references.citation: Science. 2000 Mar 17;287(5460):2032-6
references.pmid: 10720333
references.citation: Dev Biol. 2000 Apr 15;220(2):197-210
references.pmid: 10753510
references.citation: Invest Ophthalmol Vis Sci. 2001 Mar;42(3):868-74
references.pmid: 11222552
references.citation: Dev Biol. 2001 Jun 15;234(2):454-69
references.pmid: 11397013
references.citation: Prog Brain Res. 2001;131:565-77
references.pmid: 11420971
references.citation: J Struct Biol. 2001 Aug;135(2):176-84
references.pmid: 11580267
references.citation: Curr Biol. 2001 Oct 2;11(19):1481-91
references.pmid: 11591315
references.citation: Curr Biol. 2001 Oct 2;11(19):1492-502
references.pmid: 11591316
references.citation: Traffic. 2001 Dec;2(12):867-72
references.pmid: 11737824
references.citation: Dev Dyn. 2001 Dec;222(4):625-36
references.pmid: 11748831
references.citation: Annu Rev Physiol. 2002;64:899-923
references.pmid: 11826292
references.citation: Nat Rev Mol Cell Biol. 2002 Feb;3(2):94-103
references.pmid: 11836511
references.citation: Proc Natl Acad Sci U S A. 2002 Mar 19;99(6):3842-7
references.pmid: 11904436
references.citation: J Cell Biol. 2002 Apr 1;157(1):103-13
references.pmid: 11916979
references.citation: Nat Genet. 2002 Jun;31(2):150-7
references.pmid: 11992120
references.citation: Nat Genet. 2002 Jun;31(2):135-40
references.pmid: 12006978
references.citation: Development. 2002 Jul;129(13):3137-46
references.pmid: 12070089
references.citation: Development. 2002 Jul;129(14):3281-94
references.pmid: 12091300
references.citation: J Cell Sci. 1996 Jan;109 ( Pt 1):271-6
references.pmid: 8834812