CNS Department, Boehringer Ingelheim Pharma GmbH& Co. KG, Biberach an der Riss, Germany.
Department of Child and Adolescent Psychiatry/Psychotherapy, University of Ulm, Ulm, Germany.
PLoS One. 2018 Apr 26;13(4):e0196515. doi: 10.1371/journal.pone.0196515. eCollection 2018.
Motor tics are sudden, repetitive, involuntary movements representing the hallmark behaviors of the neurodevelopmental disease Tourette's syndrome (TS). The primary cause of TS remains unclear. The initial observation that dopaminergic antagonists alleviate tics led to the development of a dopaminergic theory of TS etiology which is supported by post mortem and in vivo studies indicating that non-physiological activation of the striatum could generate tics. The striatum controls movement execution through the balanced activity of dopamine receptor D1 and D2-expressing medium spiny neurons of the direct and indirect pathway, respectively. Different neurotransmitters can activate or repress striatal activity and among them, dopamine plays a major role. In this study we introduced a chronic dopaminergic alteration in juvenile rats, in order to modify the delicate balance between direct and indirect pathway. This manipulation was done in the dorsal striatum, that had been associated with tic-like movements generation in animal models. The results were movements resembling tics, which were categorized and scored according to a newly developed rating scale and were reduced by clonidine and riluzole treatment. Finally, post mortem analyses revealed altered RNA expression of dopaminergic receptors D1 and D2, suggesting an imbalanced dopaminergic regulation of medium spiny neuron activity as being causally related to the observed phenotype.
运动性抽动是一种突发性、重复性、无意识的运动,是神经发育性疾病妥瑞氏综合征(TS)的标志性行为。TS 的主要病因仍不清楚。最初观察到多巴胺拮抗剂可减轻抽动,这导致了 TS 病因的多巴胺能理论的发展,该理论得到了死后和体内研究的支持,这些研究表明纹状体的非生理性激活可能产生抽动。纹状体通过多巴胺受体 D1 和 D2 表达的中间棘神经元的直接和间接通路的平衡活动来控制运动执行,分别。不同的神经递质可以激活或抑制纹状体的活动,其中多巴胺起着主要作用。在这项研究中,我们在幼年大鼠中引入了慢性多巴胺能改变,以改变直接和间接通路之间的微妙平衡。这种操作是在背侧纹状体中进行的,背侧纹状体与动物模型中的类抽动运动的产生有关。结果是类似于抽动的运动,根据新开发的评分量表对其进行分类和评分,并通过可乐定和利鲁唑治疗减少。最后,死后分析显示多巴胺受体 D1 和 D2 的 RNA 表达改变,表明中间棘神经元活动的多巴胺能调节失衡与观察到的表型有因果关系。