molecular-genetic mapping of zebrafish mutants with variable phenotypic penetrance分子遗传学映射变量斑马鱼突变体的表型外显率.pdfVIP

Molecular-Genetic Mapping of Zebrafish Mutants with Variable Phenotypic Penetrance . . ¤ Roshan A. Jain , Marc A. Wolman , Lauren A. Schmidt, Harold A. Burgess , Michael Granato* Department of Cell and Developmental Biology, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania, United States of America Abstract Forward genetic screens in vertebrates are powerful tools to generate models relevant to human diseases, including neuropsychiatric disorders. Variability in phenotypic penetrance and expressivity is common in these disorders and behavioral mutant models, making their molecular-genetic mapping a formidable task. Using a ‘phenotyping by segregation’ strategy, we molecularly map the hypersensitive zebrafish houdini mutant despite its variable phenotypic penetrance, providing a generally applicable strategy to map zebrafish mutants with subtle phenotypes. Citation: Jain RA, Wolman MA, Schmidt LA, Burgess HA, Granato M (2011) Molecular-Genetic Mapping of Zebrafish Mutants with Variable Phenotypic Penetrance. PLoS ONE 6(10): e26510. doi:10.1371/journal.pone.0026510 Editor: Denis Dupuy, Inserm U869, France Received June 3, 2011; Accepted September 28, 2011; Published October 19, 2011 Copyright: 2011 Jain et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Funding: This work was funded by a NRSA postdoctoral fellowship (5F32NS065637–02) to R.A.J., a NRSA postdoctoral fellowship (5F32EY019434–03) to M.A.W., and a National Inst