Schnerwitzki Danny, Perry Sharn, Ivanova Anna, Caixeta Fabio V, Cramer Paul, Günther Sven, Weber Kathrin, Tafreshiha Atieh, Becker Lore, Vargas Panesso Ingrid L, Klopstock Thomas, Hrabe de Angelis Martin, Schmidt Manuela, Kullander Klas, Englert Christoph
Molecular Genetics Lab, Leibniz Institute on Aging-Fritz Lipmann Institute, Jena, Germany.
Department of Neuroscience, Uppsala University, Uppsala, Sweden.
Life Sci Alliance. 2018 Aug 16;1(4):e201800106. doi: 10.26508/lsa.201800106. eCollection 2018 Aug.
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 . 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 deletion. Locomotion analyses of mice with neuron-specific 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 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 mutants was significantly altered. These results show that the development of particular dI6 neurons depends on expression and that loss of is associated with alterations in locomotion.
运动由脊髓的神经回路协调。最近，dI6神经元被证明参与运动控制。dI6神经元的一个亚群表达威尔姆斯肿瘤抑制基因。然而，Wt1在这些细胞中的功能尚不清楚。在这里，我们旨在确定与缺失相关的脊髓行为变化和细胞改变。对具有神经元特异性缺失的小鼠进行的运动分析显示，其行走速度较慢，步频降低，步长增加。这些小鼠的前肢/后肢协调性发生了变化，同时脊髓中对侧投射丧失。具有缺失的新生儿表现出不协调的后肢运动增加，其运动神经元输出无节律且频率降低。条件性突变体发育中的脊髓中dI6、V0和V2a神经元的群体大小发生了显著改变。这些结果表明，特定dI6神经元的发育依赖于表达，并且的缺失与运动改变有关。
