Intellectual and Developmental Disabilities Research Center, Jane and Terry Semel Institute for Neuroscience & Human Behavior.
Department of Neurology, David Geffen School of Medicine, University of California Los Angeles, Los Angeles, CA 90095, USA.
Cereb Cortex. 2020 Apr 14;30(4):2372-2388. doi: 10.1093/cercor/bhz245.
Huntington's disease (HD) is a neurodegenerative disorder characterized by involuntary movements, cognitive deficits, and psychiatric disturbances. Although evidence indicates that projections from motor cortical areas play a key role in the development of dysfunctional striatal activity and motor phenotype, little is known about the changes in cortical microcircuits and their role in the development of the HD phenotype. Here we used two-photon laser-scanning microscopy to evaluate network dynamics of motor cortical neurons in layers II/III in behaving transgenic R6/2 and knock-in Q175+/- mice. Symptomatic R6/2 mice displayed increased motion manifested by a significantly greater number of motion epochs, whereas symptomatic Q175 mice displayed decreased motion. In both models, calcium transients in symptomatic mice displayed reduced amplitude, suggesting decreased bursting activity. Changes in frequency were genotype- and time-dependent; for R6/2 mice, the frequency was reduced during both motion and nonmotion, whereas in symptomatic Q175 mice, the reduction only occurred during nonmotion. In presymptomatic Q175 mice, frequency was increased during both behavioral states. Interneuronal correlation coefficients were generally decreased in both models, suggesting disrupted interneuronal communication in HD cerebral cortex. These results indicate similar and contrasting effects of the HD mutation on cortical ensemble activity depending on mouse model and disease stage.
亨廷顿病(HD)是一种神经退行性疾病,其特征为不自主运动、认知缺陷和精神障碍。虽然有证据表明,运动皮质区域的投射在纹状体活动和运动表型的功能障碍发展中起着关键作用,但对于皮质微电路的变化及其在 HD 表型发展中的作用知之甚少。在这里,我们使用双光子激光扫描显微镜来评估行为转基因 R6/2 和敲入 Q175+/- 小鼠的运动皮质神经元在 II/III 层中的网络动力学。有症状的 R6/2 小鼠表现出明显更多运动时期的增加运动,而有症状的 Q175 小鼠则表现出减少运动。在这两种模型中,有症状的小鼠的钙瞬变幅度减小,表明爆发活动减少。频率变化与基因型和时间有关;对于 R6/2 小鼠,在运动和非运动期间频率均降低,而在有症状的 Q175 小鼠中,仅在非运动期间降低。在有症状的 Q175 小鼠中,在两种行为状态下频率均增加。两种模型中的中间神经元相关系数通常降低,表明 HD 大脑皮质中中间神经元通讯中断。这些结果表明,HD 突变对皮质集合活动的影响在不同的小鼠模型和疾病阶段相似但又有所不同。