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Molecular-Genetic Mapping of Zebrafish Mutants with Variable Phenotypic Penetrance

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 for...

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Detalles Bibliográficos
Autores principales: Jain, Roshan A., Wolman, Marc A., Schmidt, Lauren A., Burgess, Harold A., Granato, Michael
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3198425/
https://www.ncbi.nlm.nih.gov/pubmed/22039502
http://dx.doi.org/10.1371/journal.pone.0026510
Descripción
Sumario: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.