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Loss of the neurodevelopmental gene Zswim6 alters striatal morphology and motor regulation.


ABSTRACT: The zinc-finger SWIM domain-containing protein 6 (ZSWIM6) is a protein of unknown function that has been associated with schizophrenia and limited educational attainment by three independent genome-wide association studies. Additionally, a putatively causal point mutation in ZSWIM6 has been identified in several cases of acromelic frontonasal dysostosis with severe intellectual disability. Despite the growing number of studies implicating ZSWIM6 as an important regulator of brain development, its role in this process has never been examined. Here, we report the generation of Zswim6 knockout mice and provide a detailed anatomical and behavioral characterization of the resulting phenotype. We show that Zswim6 is initially expressed widely during embryonic brain development but becomes restri

SUBMITTER: Tischfield DJ 

PROVIDER: S-EPMC5703052 | biostudies-literature | 2017 Jul

REPOSITORIES: biostudies-literature