Center for Neuroscience, Biosciences Division, SRI International, Menlo Park, California 94025 and Department of Neuroscience II, Research Institute of Environmental Medicine, Nagoya University, Nagoya 464-8601, Japan.
J Neurosci. 2014 May 7;34(19):6485-94. doi: 10.1523/JNEUROSCI.0080-14.2014.
γ-Hydroxybutyrate (GHB) is an approved therapeutic for the excessive sleepiness and sudden loss of muscle tone (cataplexy) characteristic of narcolepsy. The mechanism of action for these therapeutic effects is hypothesized to be GABAB receptor dependent. We evaluated the effects of chronic administration of GHB and the GABAB agonist R-baclofen (R-BAC) on arousal state and cataplexy in two models of narcolepsy: orexin/ataxin-3 (Atax) and orexin/tTA; TetO diphtheria toxin mice (DTA). Mice were implanted for EEG/EMG monitoring and dosed with GHB (150 mg/kg), R-BAC (2.8 mg/kg), or vehicle (VEH) bid for 15 d-a treatment paradigm designed to model the twice nightly GHB dosing regimen used by human narcoleptics. In both models, R-BAC increased NREM sleep time, intensity, and consolidation during the light period; wake bout duration increased and cataplexy decreased during the subsequent dark period. GHB did not increase NREM sleep consolidation or duration, although NREM delta power increased in the first hour after dosing. Cataplexy decreased from baseline in 57 and 86% of mice after GHB and R-BAC, respectively, whereas cataplexy increased in 79% of the mice after VEH. At the doses tested, R-BAC suppressed cataplexy to a greater extent than GHB. These results suggest utility of R-BAC-based therapeutics for narcolepsy.
γ-羟基丁酸(GHB)是一种已被批准的治疗药物,可用于治疗嗜睡症和猝倒(肌肉突然失去张力)的特征。其作用机制被假设为 GABA B 受体依赖性。我们评估了 GHB 和 GABA B 激动剂 R-巴氯芬(R-BAC)在两种嗜睡症模型中的慢性给药对觉醒状态和猝倒的影响:食欲素/ataxin-3(Atax)和食欲素/tTA; Orexin/tTA;TetO 白喉毒素小鼠(DTA)。在 EEG/EMG 监测下植入了这些小鼠,并给予 GHB(150mg/kg)、R-BAC(2.8mg/kg)或对照剂(VEH)bid,共 15d,这一治疗方案旨在模拟人类嗜睡症患者每晚两次给予 GHB 的方案。在这两种模型中,R-BAC 增加了光照期间 NREM 睡眠时间、强度和巩固程度;在随后的黑暗期,觉醒持续时间延长,猝倒减少。GHB 并未增加 NREM 睡眠的巩固或持续时间,尽管在给药后 1 小时内 NREM 德尔塔功率增加。在 GHB 和 R-BAC 治疗后,分别有 57%和 86%的小鼠猝倒减少到基线以下,而 79%的小鼠在 VEH 治疗后猝倒增加。在测试的剂量下,R-BAC 抑制猝倒的程度比 GHB 更大。这些结果表明,基于 R-BAC 的治疗方法对嗜睡症具有应用价值。