Systematic genome-wide mutagenesis screens for embryonic phenotypes have been instrumental in the understanding of invertebrate and plant development. Here, we report the results from the first application of such a large-scale genetic screening to vertebrate development. Male zebrafish were mutagenized with N-ethyl N-nitrosourea to induce mutations in spermatogonial cells at an average specific locus rate of one in 651 mutagenized genomes. Mutations were transmitted to the F1 generation, and 2205 F2 families were raised. F3 embryos from sibling crosses within the F2 families were screened for developmental abnormalities. A total of 2337 mutagenized genomes were analyzed, and 2383 mutations resulting in abnormal embryonic and early larval phenotypes were identified. The phenotypes of 695 mutants indicated involvement of the identified loci in specific aspects of embryogenesis. These mutations were maintained for further characterization and were classified into categories according to their phenotypes. The analyses and genetic complementation of mutations from several categories are reported in separate manuscripts. Mutations affecting pigmentation, motility, muscle and body shape have not been extensively analyzed and are listed here. A total of 331 mutations were tested for allelism within their respective categories. This defined 220 genetic loci with on average 1.5 alleles per locus. For about two-thirds of all loci only one allele was isolated. Therefore it is not possible to give a reliable estimate on the degree of saturation reached in our screen; however, the number of genes that can mutate to visible embryonic and early larval phenotypes in zebrafish is expected to be several-fold larger than the one for which we have observed mutant alleles during the screen. This screen demonstrates that mutations affecting a variety of developmental processes can be efficiently recovered from zebrafish.
A genetic screen for mutations affecting embryogenesis in zebrafish
W. Driever, L. Solnica-Krezel, A.F. Schier, S.C. Neuhauss, J. Malicki, D.L. Stemple, D.Y. Stainier, F. Zwartkruis, S. Abdelilah, Z. Rangini, J. Belak, C. Boggs; A genetic screen for mutations affecting embryogenesis in zebrafish. Development 1 December 1996; 123 (1): 37–46. doi: https://doi.org/10.1242/dev.123.1.37
Download citation file:
Advertisement
Cited by
Pathway to Independence programme

We’re excited to announce our new Pathway to Independence programme, aimed at supporting postdocs as they go on the job market. Find out more about the scheme in our Editorial.
Call for papers: Metabolic and Nutritional Control of Development and Regeneration

We are welcoming submissions for our next special issue, which will focus on metabolic and nutritional control of development and regeneration. Submission deadline: 15 May 2023.
Webinar: Increasing the visibility and impact of your research
-HUBSwebinar.jpg?versionId=4486)
Would you like to increase the visibility and impact of your research and raise your profile internationally? If so, register for the very practical webinar we are running in association with HUBS on 23 February 2023.
Transitions in development: Daniel Grimes

Daniel Grimes’s lab studies the consequences of ciliary mutations, including left-right patterning defects and scoliosis. We interviewed Daniel to find out more about his career path, his experience of becoming a group leader and the influence of Jurassic Park.
Preprints in Development
(update)-InPreprints.png?versionId=4486)
As part of our efforts to support the use of preprints and help curate the preprint literature, we are delighted to launch a new article type: ‘In preprints’. These pieces will discuss one or more recent preprints and place them in a broader context.