Boulanger Talia, Pigeon Pascale, Crawford Stephen
Talia Boulanger, LLC, Westford, MA, USA.
Takeda Development Center Americas, Cambridge, MA, USA.
Sleep Adv. 2024 Aug 16;5(1):zpae059. doi: 10.1093/sleepadvances/zpae059. eCollection 2024.
Idiopathic hypersomnia (IH) is a rare neurological sleep disorder, characterized by excessive daytime sleepiness despite normal sleep duration, that can significantly impact patient's lives. The burden of IH goes beyond excessive daytime sleepiness, pervading all aspects of everyday life. Characteristic and burdensome symptoms of IH include sleep inertia/drunkenness, long sleep duration, and daytime cognitive dysfunction. This systematic review assessed current knowledge regarding IH diagnostic challenges and burden of illness. Literature searches for original epidemiological, clinical, humanistic, or economic research relevant to IH published between 2012 and 2022 in MEDLINE, Embase, Cochrane, gray literature (diagnostic criteria and treatment guidelines), conferences (2019-2022), and clinical trial databases yielded 97 articles. Findings indicate that IH remains a poorly defined diagnosis of exclusion that is difficult to distinguish from narcolepsy type 2 because of symptom overlap and inadequacies of objective testing. Consequently, individuals with IH endure diagnostic delays of up to 9 years. The economic burden of IH has not been characterized to any appreciable extent. Pharmacological treatment options can improve symptoms and functional status, but rarely restores normal levels of functioning. These findings highlight the need to reclassify central disorders of hypersomnolence. Further collaboration is now required between research groups to identify and validate objective markers to help redefine diagnostic criteria for IH. This would move IH into a position that could benefit from future targeted therapeutic interventions. The study was funded by Takeda Development Center Americas, Inc.
特发性嗜睡症(IH)是一种罕见的神经睡眠障碍,其特征是尽管睡眠时间正常,但白天仍过度嗜睡,这会对患者的生活产生重大影响。IH的负担不仅限于白天过度嗜睡,还渗透到日常生活的方方面面。IH的典型且繁重的症状包括睡眠惯性/醉酒感、睡眠时间长以及白天认知功能障碍。本系统综述评估了有关IH诊断挑战和疾病负担的现有知识。在MEDLINE、Embase、Cochrane、灰色文献(诊断标准和治疗指南)、会议(2019 - 2022年)以及临床试验数据库中,检索2012年至2022年间发表的与IH相关的原始流行病学、临床、人文或经济研究,共获得97篇文章。研究结果表明,IH仍然是一种定义不明确的排除性诊断,由于症状重叠和客观测试的不足,很难与2型发作性睡病区分开来。因此,患有IH的个体要忍受长达9年的诊断延迟。IH的经济负担在很大程度上尚未得到描述。药物治疗选择可以改善症状和功能状态,但很少能恢复正常功能水平。这些发现凸显了对中枢性过度嗜睡症进行重新分类的必要性。现在研究小组之间需要进一步合作,以识别和验证客观标志物,帮助重新定义IH的诊断标准。这将使IH能够受益于未来有针对性的治疗干预。该研究由美国武田开发中心资助。