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Molecular-genetic mapping of zebrafish mutants with variable phenotypic penetrance.


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.

SUBMITTER: Jain RA 

PROVIDER: S-EPMC3198425 | biostudies-literature | 2011

REPOSITORIES: biostudies-literature

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Molecular-genetic mapping of zebrafish mutants with variable phenotypic penetrance.

Jain Roshan A RA   Wolman Marc A MA   Schmidt Lauren A LA   Burgess Harold A HA   Granato Michael M  

PloS one 20111019 10


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  ...[more]

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