Suppr超能文献

睡眠障碍性 Kleine-Levin 综合征(KLS)加入了与产科并发症相关的多基因脑疾病列表。

Sleep Disorder Kleine-Levin Syndrome (KLS) Joins the List of Polygenic Brain Disorders Associated with Obstetric Complications.

机构信息

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.

出版信息

Cell Mol Neurobiol. 2023 Oct;43(7):3393-3403. doi: 10.1007/s10571-023-01391-z. Epub 2023 Aug 9.

Abstract

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.

摘要

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。

相似文献

3
Pharmacological treatment for Kleine-Levin syndrome.克莱恩-莱文综合征的药物治疗。
Cochrane Database Syst Rev. 2016 May 6;2016(5):CD006685. doi: 10.1002/14651858.CD006685.pub4.
4
Kleine-Levin syndrome is associated with LMOD3 variants.克莱恩-莱文综合征与 LMOD3 变异有关。
J Sleep Res. 2019 Jun;28(3):e12718. doi: 10.1111/jsr.12718. Epub 2018 Jun 19.
5
Pharmacological treatment for Kleine-Levin syndrome.克莱恩-莱文综合征的药物治疗
Cochrane Database Syst Rev. 2013 Aug 14(8):CD006685. doi: 10.1002/14651858.CD006685.pub3.
9
Kleine-Levin Syndrome.克莱恩-莱文综合征。
Paediatr Respir Rev. 2018 Jan;25:9-13. doi: 10.1016/j.prrv.2016.12.004. Epub 2016 Dec 23.
10
Idiopathic hypersomnia and Kleine-Levin syndrome.特发性嗜睡症和 Kleine-Levin 综合征。
Rev Neurol (Paris). 2023 Oct;179(7):741-754. doi: 10.1016/j.neurol.2023.08.010. Epub 2023 Sep 6.

本文引用的文献

1
Genetics and epigenetics of rare hypersomnia.罕见嗜睡症的遗传学和表观遗传学。
Trends Genet. 2023 May;39(5):415-429. doi: 10.1016/j.tig.2023.02.003. Epub 2023 Feb 24.
2
An Update on Kleine-Levin Syndrome.克莱恩-莱文综合征最新进展
Curr Sleep Med Rep. 2023;9(1):35-44. doi: 10.1007/s40675-022-00246-1. Epub 2022 Dec 27.
5
LMOD3 gene variant in familial periodic hypersomnolence.家族性周期性嗜睡症中的LMOD3基因变异
Sleep Med. 2022 Mar;91:105-108. doi: 10.1016/j.sleep.2022.02.019. Epub 2022 Feb 28.
9
Regulation of TRANK1 by GSK-3 in the brain: unexpected interactions.GSK-3对大脑中TRANK1的调控:意外的相互作用。
Mol Psychiatry. 2021 Nov;26(11):6109-6111. doi: 10.1038/s41380-021-01120-2. Epub 2021 Apr 30.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验