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

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Schnerwitzki, D, Perry, S, Ivanova, A, Caixeta, FV, Cramer, P, Gunther, S, Weber, K, Tafreshiha, A, Becker, L, Vargas Panesso, IL, Klopstock, T, Hrabe de Angelis, M, Schmidt, M, Kullander, K and Englert, C 2018 , 'Neuron-specific inactivation of Wt1 alters locomotion in mice and changes interneuron composition in the spinal cord' , Life science alliance, vol. 1, no. 4 , pp. 1-15 , doi: 10.26508/lsa.201800106.

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Abstract

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.

Item Type: Article
Authors/Creators:Schnerwitzki, D and Perry, S and Ivanova, A and Caixeta, FV and Cramer, P and Gunther, S and Weber, K and Tafreshiha, A and Becker, L and Vargas Panesso, IL and Klopstock, T and Hrabe de Angelis, M and Schmidt, M and Kullander, K and Englert, C
Keywords: CPG; WT1; central pattern generator; interneuron; locomotion
Journal or Publication Title: Life science alliance
Publisher: Life Science Alliance
ISSN: 2575-1077
DOI / ID Number: 10.26508/lsa.201800106
Copyright Information:

Copyright 2018 Schnerwitzki et al. Licensed under Creative Commons Attribution 4.0 International (CC BY 4.0) https://creativecommons.org/licenses/by/4.0/

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