International Institute for Integrative Sleep Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-8575, Japan.
International Institute for Integrative Sleep Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-8575, Japan
Proc Natl Acad Sci U S A. 2017 May 30;114(22):5731-5736. doi: 10.1073/pnas.1700499114. Epub 2017 May 15.
Narcolepsy-cataplexy is a debilitating disorder of sleep/wakefulness caused by a loss of orexin-producing neurons in the lateroposterior hypothalamus. Genetic or pharmacologic orexin replacement ameliorates symptoms in mouse models of narcolepsy-cataplexy. We have recently discovered a potent, nonpeptide OX2R-selective agonist, YNT-185. This study validates the pharmacological activity of this compound in OX2R-transfected cells and in OX2R-expressing neurons in brain slice preparations. Intraperitoneal, and intracerebroventricular, administration of YNT-185 suppressed cataplexy-like episodes in orexin knockout and orexin neuron-ablated mice, but not in orexin receptor-deficient mice. Peripherally administered YNT-185 also promotes wakefulness without affecting body temperature in wild-type mice. Further, there was no immediate rebound sleep after YNT-185 administration in active phase in wild-type and orexin-deficient mice. No desensitization was observed after repeated administration of YNT-185 with respect to the suppression of cataplexy-like episodes. These results provide a proof-of-concept for a mechanistic therapy of narcolepsy-cataplexy by OX2R agonists.
发作性睡病伴猝倒症是一种由下丘脑外侧后部的食欲素神经元缺失引起的睡眠/觉醒障碍性疾病。在发作性睡病伴猝倒症的小鼠模型中,遗传或药理学食欲素替代疗法可改善症状。我们最近发现了一种有效的、非肽类 OX2R 选择性激动剂 YNT-185。本研究验证了该化合物在 OX2R 转染细胞和脑片制备中 OX2R 表达神经元中的药理学活性。腹腔内和侧脑室给予 YNT-185 可抑制食欲素敲除和食欲素神经元消融小鼠的猝倒样发作,但对食欲素受体缺陷小鼠无效。外周给予 YNT-185 也可促进觉醒,而不影响野生型小鼠的体温。此外,在野生型和食欲素缺陷小鼠的活动期,给予 YNT-185 后没有立即出现睡眠反弹。重复给予 YNT-185 后,对猝倒样发作的抑制作用没有出现脱敏现象。这些结果为 OX2R 激动剂治疗发作性睡病伴猝倒症提供了一个概念验证。