RT Journal Article SR Electronic T1 Neuron-specific inactivation of Wt1 alters locomotion in mice and changes interneuron composition in the spinal cord JF Life Science Alliance JO Life Sci. Alliance FD Life Science Alliance LLC SP e201800106 DO 10.26508/lsa.201800106 VO 1 IS 4 A1 Danny Schnerwitzki A1 Sharn Perry A1 Anna Ivanova A1 Fabio V Caixeta A1 Paul Cramer A1 Sven Günther A1 Kathrin Weber A1 Atieh Tafreshiha A1 Lore Becker A1 Ingrid L Vargas Panesso A1 Thomas Klopstock A1 Martin Hrabe de Angelis A1 Manuela Schmidt A1 Klas Kullander A1 Christoph Englert YR 2018 UL https://www.life-science-alliance.org/content/1/4/e201800106.abstract AB 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.