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Neuron-specific inactivation of Wt1 alters locomotion in mice and changes interneuron composition in the spinal cord
Fritz Lipmann Inst, Leibniz Inst Aging, Mol Genet Lab, Jena, Germany.
Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Neuroscience, Developmental Genetics.
Fritz Lipmann Inst, Leibniz Inst Aging, Mol Genet Lab, Jena, Germany.
Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Neuroscience, Developmental Genetics.
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2018 (English)In: LIFE SCIENCE ALLIANCE, ISSN 2575-1077, Vol. 1, no 4, article id e201800106Article in journal (Refereed) Published
Abstract [en]

Locomotion is coordinated by neuronal circuits of the spinal cord. Recently, dI6 neurons were shown to participate in the control of locomotion. A subpopulation of dI6 neurons expresses the Wilms tumor suppressor gene Wt1. However, the function of Wt1 in these cells is not understood. Here, we aimed to identify behavioral changes and cellular alterations in the spinal cord associated with Wt1 deletion. Locomotion analyses of mice with neuron-specific Wt1 deletion revealed a slower walk with a decreased stride frequency and an increased stride length. These mice showed changes in their fore-/hindlimb coordination, which were accompanied by a loss of contralateral projections in the spinal cord. Neonates with Wt1 deletion displayed an increase in uncoordinated hindlimb movements and their motor neuron output was arrhythmic with a decreased frequency. The population size of dI6, V0, and V2a neurons in the developing spinal cord of conditional Wt1 mutants was significantly altered. These results show that the development of particular dI6 neurons depends on Wt1 expression and that loss of Wt1 is associated with alterations in locomotion.

Place, publisher, year, edition, pages
2018. Vol. 1, no 4, article id e201800106
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Neurosciences
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URN: urn:nbn:se:uu:diva-377292DOI: 10.26508/lsa.201800106ISI: 000457327700010PubMedID: 30456369OAI: oai:DiVA.org:uu-377292DiVA, id: diva2:1289554
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The Swedish Brain FoundationAvailable from: 2019-02-18 Created: 2019-02-18 Last updated: 2019-02-18Bibliographically approved

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