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Life Sci Alliance. 2018 Aug 16;1(4):e201800106. doi: 10.26508/lsa.201800106. eCollection 2018 Aug.

Neuron-specific inactivation of Wt1 alters locomotion in mice and changes interneuron composition in the spinal cord.

Author information

Molecular Genetics Lab, Leibniz Institute on Aging-Fritz Lipmann Institute, Jena, Germany.
Department of Neuroscience, Uppsala University, Uppsala, Sweden.
German Mouse Clinic, Institute of Experimental Genetics, Helmholtz Zentrum München, German Research Center for Environmental Health, Neuherberg, Germany.
Department of Neurology, Friedrich-Baur-Institut, Ludwig Maximilian University Munich, Munich, Germany.
German Center for Neurodegenerative Diseases, Munich, Germany.
Munich Cluster for Systems Neurology, Adolf-Butenandt-Institut, Ludwig Maximilian University Munich, Munich, Germany.
German Center for Vertigo and Balance Disorders, University Hospital Munich, Campus Grosshadern, Munich, Germany.
Chair of Experimental Genetics, School of Life Science Weihenstephan, Technical University of Munich, Freising, Germany.
German Center for Diabetes Research, Neuherberg, Germany.
Institute of Systematic Zoology and Evolutionary Biology with Phyletic Museum, Friedrich Schiller University Jena, Jena, Germany.
Institute of Biochemistry and Biophysics, Friedrich-Schiller-University Jena, Jena, Germany.


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.

Conflict of interest statement

The authors declare that they have no conflict of interest.

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