Florida Atlantic University (FAU), CE Schmidt College of Medicine, Boca Raton, FL, USA.
Dept. Clinical Neurosciences, CE Schmidt College of Medicine, Florida Atlantic University (FAU), 777 Glades Road, Boca Raton, FL, 33431, USA.
Cell Mol Neurobiol. 2023 Oct;43(7):3393-3403. doi: 10.1007/s10571-023-01391-z. Epub 2023 Aug 9.
Kleine-Levin Syndrome is a rare neurological disorder with onset typically during adolescence that is characterized by recurrent episodes of hypersomnia, behavioral changes, and cognitive abnormalities, in the absence of structural changes in neuroimaging. As for many functional brain disorders, the exact disease mechanism in Kleine-Levin Syndrome is presently unknown, preventing the development of specific treatment approaches or protective measures. Here we review the pathophysiology and genetics of this functional brain disorder and then present a specific working hypothesis. A neurodevelopmental mechanism has been suspected based on associations with obstetric complications. Recent studies have focused on genetic factors whereby the first genome-wide association study (GWAS) in Kleine-Levin Syndrome has defined a linkage at the TRANK1 locus. A Gene x Environment interaction model involving obstetric complications was proposed based on concepts developed for other functional brain disorders. To stimulate future research, we here performed annotations of the genes under consideration for Kleine-Levin Syndrome in relation to factors expected to be associated with obstetric complications. Annotations used data-mining of gene/protein lists related to for hypoxia, ischemia, and vascular factors and targeted literature searches. Tentative links for TRANK1, four additional genes in the TRANK1 locus, and LMOD3-LMO2 are described. Protein interaction data for TRANK1 indicate links to CBX2, CBX4, and KDM3A, that in turn can be tied to hypoxia. Taken together, the neurological sleep disorder, Kleine-Levin Syndrome, shows genetic and mechanistic overlap with well analyzed brain disorders such as schizophrenia, autism spectrum disorder and ADHD in which polygenic predisposition interacts with external events during brain development, including obstetric complications.
克莱恩-莱文综合征是一种罕见的神经紊乱疾病,通常在青少年时期发病,其特征是反复发作的过度嗜睡、行为改变和认知异常,但神经影像学检查没有结构变化。由于许多功能性脑疾病的原因尚不清楚,目前尚不清楚克莱恩-莱文综合征的确切发病机制,这阻碍了特定治疗方法或保护措施的发展。在这里,我们回顾了这种功能性脑疾病的病理生理学和遗传学,然后提出了一个具体的工作假设。基于与产科并发症的关联,怀疑存在神经发育机制。最近的研究集中在遗传因素上,第一项克莱恩-莱文综合征的全基因组关联研究(GWAS)已经确定了 TRANK1 基因座的连锁。基于其他功能性脑疾病的概念,提出了一个基因与环境相互作用的模型,其中包括产科并发症。为了激发未来的研究,我们对与产科并发症相关的克莱恩-莱文综合征相关基因进行了注释。注释使用了与缺氧、缺血和血管因素相关的基因/蛋白质列表的数据挖掘,并进行了有针对性的文献搜索。描述了 TRANK1、TRANK1 基因座中的另外四个基因和 LMOD3-LMO2 的暂定联系。TRANK1 的蛋白质相互作用数据表明与 CBX2、CBX4 和 KDM3A 有联系,而 CBX4 和 KDM3A 又可以与缺氧联系起来。总的来说,神经睡眠障碍克莱恩-莱文综合征在遗传和机制上与经过充分分析的脑疾病(如精神分裂症、自闭症谱系障碍和注意力缺陷多动障碍)重叠,这些疾病的多基因易感性与大脑发育过程中的外部事件(包括产科并发症)相互作用。