Identification of zebrafish insertional mutants with defects in visual system development and function

被引:114
作者
Gross, JM
Perkins, BD
Amsterdam, A
Egaña, A
Darland, T
Matsui, JI
Sciascia, S
Hopkins, N
Dowling, JE
机构
[1] Harvard Univ, Dept Mol & Cellular Biol, Cambridge, MA 02138 USA
[2] MIT, Ctr Canc Res, Cambridge, MA 02139 USA
[3] MIT, Dept Biol, Cambridge, MA 02139 USA
关键词
D O I
10.1534/genetics.104.039727
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
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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页码:245 / 261
页数:17
相关论文
共 67 条
[1]   Insertional mutagenesis in zebrafish identifies two novel genes, pescadillo and dead eye, essential for embryonic development [J].
Allende, ML ;
Amsterdam, A ;
Becker, T ;
Kawakami, K ;
Gaiano, N ;
Hopkins, N .
GENES & DEVELOPMENT, 1996, 10 (24) :3141-3155
[2]   Genetic diseases of acid-base transporters [J].
Alper, SL .
ANNUAL REVIEW OF PHYSIOLOGY, 2002, 64 :899-923
[3]   Identification of 315 genes essential for early zebrafish development [J].
Amsterdam, A ;
Nissen, RM ;
Sun, ZX ;
Swindell, EC ;
Farrington, S ;
Hopkins, N .
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 2004, 101 (35) :12792-12797
[4]   Insertional mutagenesis in zebrafish [J].
Amsterdam, A .
DEVELOPMENTAL DYNAMICS, 2003, 228 (03) :523-534
[5]   A large-scale insertional mutagenesis screen in zebrafish [J].
Amsterdam, A ;
Burgess, S ;
Golling, G ;
Chen, WB ;
Sun, ZX ;
Townsend, K ;
Farrington, S ;
Haldi, M ;
Hopkins, N .
GENES & DEVELOPMENT, 1999, 13 (20) :2713-2724
[6]  
[Anonymous], 1995, ZEBRAFISH BOOK
[7]  
Becker TS, 1998, DEVELOPMENT, V125, P4369
[8]   Inhibition of the ATP-driven proton pump in RPE lysosomes by the major lipofuscin fluorophore A2-E may contribute to the pathogenesis of age-related macular degeneration [J].
Bergmann, M ;
Schütt, F ;
Holz, FG ;
Kopitz, J .
FASEB JOURNAL, 2004, 18 (01) :562-+
[9]   A BEHAVIORAL SCREEN FOR ISOLATING ZEBRAFISH MUTANTS WITH VISUAL-SYSTEM DEFECTS [J].
BROCKERHOFF, SE ;
HURLEY, JB ;
JANSSENBIENHOLD, U ;
NEUHAUSS, SCF ;
DRIEVER, W ;
DOWLING, JE .
PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 1995, 92 (23) :10545-10549
[10]   Zebrafish retinal mutants [J].
Brockerhoff, SE ;
Dowling, JE ;
Hurley, JB .
VISION RESEARCH, 1998, 38 (10) :1335-1339