Lebreton Fanny, Cayzac Sebastien, Pietropaolo Susanna, Jeantet Yannick, Cho Yoon H
Institut de Neurosciences Cognitives et Intégratives d'Aquitaine, CNRS UMR 5287, Bat B2-Avenue des Facultés, 33405 Talence Cedex, France; Institut de Neurosciences Cognitives et Intégratives d'Aquitaine, CNRS UMR 5287, Bat B2-Avenue des Facultés, 33405 Talence Cedex, France.
PLoS One. 2015 May 12;10(5):e0126972. doi: 10.1371/journal.pone.0126972. eCollection 2015.
In hereditary neurodegenerative Huntington's disease (HD), there exists a growing consideration that sleep and circadian dysregulations may be important symptoms. It is not known, however, whether sleep abnormalities contribute to other behavioral deficits in HD patients and mouse models. To determine the precise chronology for sleep physiology alterations and other sensory, motor, psychiatric and cognitive symptoms of HD, the same R6/1 HD transgenics and their wild-type littermates were recorded monthly for sleep electroencephalogram (EEG) together with a wide range of behavioral tests according to a longitudinal plan. We found an early and progressive deterioration of both sleep architecture and EEG brain rhythms in R6/1 mice, which are correlated timely with their spatial working memory impairments. Sleep fragmentation and memory impairments were accompanied by the loss of delta (1-4 Hz) power in the transgenic mice, the magnitude of which increased with age and disease progression. These precocious sleep and cognitive impairments were followed by deficits in social behavior, sensory and motor abilities. Our data confirm the existence and importance of sleep physiology alterations in the widely used R6/1 mouse line and highlight their precedence over other plethoric phenotypic changes. The brainwave abnormalities, may represent a novel biomarker and point to innovative therapeutic interventions against HD.
在遗传性神经退行性疾病亨廷顿舞蹈症(HD)中,人们越来越多地认为睡眠和昼夜节律失调可能是重要症状。然而,目前尚不清楚睡眠异常是否会导致HD患者和小鼠模型出现其他行为缺陷。为了确定HD患者睡眠生理改变以及其他感觉、运动、精神和认知症状的精确时间顺序,我们按照纵向计划,每月对同一批R6/1 HD转基因小鼠及其野生型同窝小鼠进行睡眠脑电图(EEG)记录,并进行广泛的行为测试。我们发现,R6/1小鼠的睡眠结构和EEG脑节律均出现早期且渐进性的恶化,这与它们的空间工作记忆损伤及时相关。转基因小鼠的睡眠碎片化和记忆损伤伴随着δ波(1-4Hz)功率的丧失,其幅度随年龄和疾病进展而增加。这些早熟的睡眠和认知损伤之后是社交行为、感觉和运动能力的缺陷。我们的数据证实了广泛使用的R6/1小鼠品系中存在睡眠生理改变及其重要性,并强调它们比其他众多表型变化更早出现。脑电波异常可能代表一种新的生物标志物,并指向针对HD的创新治疗干预措施。