Development, Vol 123, Issue 1 1-36, Copyright © 1996 by Company of Biologists

JOURNAL ARTICLES

The identification of genes with unique and essential functions in the development of the zebrafish, Danio rerio

P Haffter, M Granato, M Brand, MC Mullins, M Hammerschmidt, DA Kane, J Odenthal, FJ van Eeden, YJ Jiang, CP Heisenberg, RN Kelsh, M Furutani-Seiki, E Vogelsang, D Beuchle, U Schach, C Fabian and C Nusslein-Volhard
Max-Planck-Institut fur Entwicklungsbiologie, Abteilung Genetik, Tubingen, Germany.

In a large-scale screen, we isolated mutants displaying a specific visible phenotype in embryos or early larvae of the zebrafish, Danio rerio. Males were mutagenized with ethylnitrosourea (ENU) and F2 families of single pair matings between sibling F1 fish, heterozygous for a mutagenized genome, were raised. Egg lays were obtained from several crosses between F2 siblings, resulting in scoring of 3857 mutagenized genomes. F3 progeny were scored at the second, third and sixth day of development, using a stereomicroscope. In a subsequent screen, fixed embryos were analyzed for correct retinotectal projection. A total of 4264 mutants were identified. Two thirds of the mutants displaying rather general abnormalities were eventually discarded. We kept and characterized 1163 mutants. In complementation crosses performed between mutants with similar phenotypes, 894 mutants have been assigned to 372 genes. The average allele frequency is 2.4. We identified genes involved in early development, notochord, brain, spinal cord, somites, muscles, heart, circulation, blood, skin, fin, eye, otic vesicle, jaw and branchial arches, pigment pattern, pigment formation, gut, liver, motility and touch response. Our collection contains alleles of almost all previously described zebrafish mutants. From the allele frequencies and other considerations we estimate that the 372 genes defined by the mutants probably represent more than half of all genes that could have been discovered using the criteria of our screen. Here we give an overview of the spectrum of mutant phenotypes obtained, and discuss the limits and the potentials of a genetic saturation screen in the zebrafish.
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				E. Shafizadeh, B. H. Paw, H. Foott, E. C. Liao, B. A. Barut, J. J. Cope, L. I. Zon, and S. Lin Characterization of zebrafish merlot/chablis as non-mammalian vertebrate models for severe congenital anemia due to protein 4.1 deficiency Development, March 11, 2003; 129(18): 4359 - 4370. [Abstract] [Full Text] [PDF]

				M. J. Parsons, I. Campos, E. M. A. Hirst, and D. L. Stemple Removal of dystroglycan causes severe muscular dystrophy in zebrafish embryos Development, March 9, 2003; 129(14): 3505 - 3512. [Abstract] [Full Text] [PDF]

				S. Burgess, G. Reim, W. Chen, N. Hopkins, and M. Brand The zebrafish spiel-ohne-grenzen (spg) gene encodes the POU domain protein Pou2 related to mammalian Oct4 and is essential for formation of the midbrain and hindbrain, and for pre-gastrula morphogenesis Development, March 4, 2003; 129(4): 905 - 916. [Abstract] [Full Text] [PDF]

				O. Biehlmaier, S. C. F. Neuhauss, and K. Kohler Double Cone Dystrophy and RPE Degeneration in the Retina of the Zebrafish gnn Mutant Invest. Ophthalmol. Vis. Sci., March 1, 2003; 44(3): 1287 - 1298. [Abstract] [Full Text] [PDF]

				J. Lo, S. Lee, M. Xu, F. Liu, H. Ruan, A. Eun, Y. He, W. Ma, W. Wang, Z. Wen, et al. 15,000 Unique Zebrafish EST Clusters and Their Future Use in Microarray for Profiling Gene Expression Patterns During Embryogenesis Genome Res., March 1, 2003; 13(3): 455 - 466. [Abstract] [Full Text] [PDF]

				J. F. Rawls, M. R. Frieda, A. R. McAdow, J. P. Gross, C. M. Clayton, C. K. Heyen, and S. L. Johnson Coupled Mutagenesis Screens and Genetic Mapping in Zebrafish Genetics, March 1, 2003; 163(3): 997 - 1009. [Abstract] [Full Text] [PDF]

				D. M. Langenau, D. Traver, A. A. Ferrando, J. L. Kutok, J. C. Aster, J. P. Kanki, S. Lin, E. Prochownik, N. S. Trede, L. I. Zon, et al. Myc-Induced T Cell Leukemia in Transgenic Zebrafish Science, February 7, 2003; 299(5608): 887 - 890. [Abstract] [Full Text] [PDF]

				J. M. Spitsbergen and M. L. Kent The State of the Art of the Zebrafish Model for Toxicology and Toxicologic Pathology Research--Advantages and Current Limitations Toxicol Pathol, January 1, 2003; 31(1_suppl): 62 - 87. [Abstract] [PDF]

				A. Donovan, A. Brownlie, M. O. Dorschner, Y. Zhou, S. J. Pratt, B. H. Paw, R. B. Phillips, C. Thisse, B. Thisse, and L. I. Zon The zebrafish mutant gene chardonnay (cdy) encodes divalent metal transporter 1 (DMT1) Blood, December 15, 2002; 100(13): 4655 - 4659. [Abstract] [Full Text] [PDF]

				H. L. Stickney, J. Schmutz, I. G. Woods, C. C. Holtzer, M. C. Dickson, P. D. Kelly, R. M. Myers, and W. S. Talbot Rapid Mapping of Zebrafish Mutations With SNPs and Oligonucleotide Microarrays Genome Res., December 1, 2002; 12(12): 1929 - 1934. [Abstract] [Full Text] [PDF]

				B. Ciruna, G. Weidinger, H. Knaut, B. Thisse, C. Thisse, E. Raz, and A. F. Schier Production of maternal-zygotic mutant zebrafish by germ-line replacement PNAS, November 12, 2002; 99(23): 14919 - 14924. [Abstract] [Full Text] [PDF]

				S. M. Prescott and H. J. Yost The COXes of Danio: From mechanistic model to experimental therapeutics PNAS, July 9, 2002; 99(14): 9084 - 9086. [Full Text] [PDF]