PT  - JOURNAL ARTICLE
AU  - Schnerwitzki, Danny
AU  - Perry, Sharn
AU  - Ivanova, Anna
AU  - Caixeta, Fabio V
AU  - Cramer, Paul
AU  - Günther, Sven
AU  - Weber, Kathrin
AU  - Tafreshiha, Atieh
AU  - Becker, Lore
AU  - Vargas Panesso, Ingrid L
AU  - Klopstock, Thomas
AU  - Hrabe de Angelis, Martin
AU  - Schmidt, Manuela
AU  - Kullander, Klas
AU  - Englert, Christoph
TI  - Neuron-specific inactivation of <em>Wt1</em> alters locomotion in mice and changes interneuron composition in the spinal cord
AID  - 10.26508/lsa.201800106
DP  - 2018 Aug 01
TA  - Life Science Alliance
PG  - e201800106
VI  - 1
IP  - 4
4099  - http://www.life-science-alliance.org/content/1/4/e201800106.short
4100  - http://www.life-science-alliance.org/content/1/4/e201800106.full
SO  - Life Sci. Alliance2018 Aug 01; 1
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.