发作性睡病1型脑脊液循环游离线粒体DNA水平升高。

IF 4.5 Q1 CLINICAL NEUROLOGY
Brain communications Pub Date : 2025-04-17 eCollection Date: 2025-01-01 DOI:10.1093/braincomms/fcaf125
Monica Moresco, Concetta Valentina Tropeano, Martina Romagnoli, Giulia Neccia, Alessandro Rapone, Fabio Pizza, Stefano Vandi, Emmanuel Mignot, Alessandra Maresca, Valerio Carelli, Giuseppe Plazzi
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引用次数: 0

摘要

1型嗜睡症(NT1)是一种罕见的神经系统疾病,以白天过度嗜睡和猝厥为特征,被认为是由针对下丘脑下丘脑分泌下丘脑分泌素的神经元的自身免疫过程引起的。为了为后一种假设提供线索,我们研究了46例脑脊液下丘脑分泌素-1低的NT1患者与32例对照组相比,脑脊液(CSF)中循环无细胞线粒体DNA (ccf-mtDNA)水平,这是神经退行性变、神经炎症或免疫激活的可能生物标志物。我们发现,与对照组相比,NT1患者的ccf-mtDNA水平显著升高,这与脑脊液下丘脑分泌素-1浓度呈负相关。此外,在睡眠开始的快速眼动期次数增加的患者中观察到较高的ccf-mtDNA水平。这些观察结果表明,ccf-mtDNA水平的升高与脑脊液下丘脑分泌素-1浓度的降低相关,从而导致睡眠结构的更大改变。此外,脑脊液中的细胞因子谱显示NT1患者中白细胞介素6和白细胞介素18的显著变化,表明活动性神经炎症过程可能与ccf-mtDNA释放有关,从而指向NT1中特定的炎症特征。这些发现暗示了NT1中潜在的线粒体功能障碍和神经炎症。需要进一步的研究来阐明潜在的机制以及这如何反映在治疗中。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Elevated circulating cell-free mitochondrial DNA level in cerebrospinal fluid of narcolepsy type 1.

Narcolepsy type 1 (NT1) is a rare neurological disorder characterized by excessive daytime sleepiness and cataplexy, thought to result from an autoimmune process targeting the hypothalamic hypocretin-producing neurons. Aiming to add clues to the latter hypothesis, we investigated circulating cell-free mitochondrial DNA (ccf-mtDNA) levels in cerebrospinal fluid (CSF), a possible biomarker for neurodegeneration, neuroinflammation or immune activation, from 46 NT1 patients with low CSF hypocretin-1, compared with 32 controls. We found significantly increased ccf-mtDNA levels in NT1 patients compared with controls, which negatively correlated with CSF hypocretin-1 concentrations. Additionally, higher ccf-mtDNA levels were observed in patients with elevated number of sleep onset rapid eye movement periods. These observations imply that increased levels of ccf-mtDNA associate with reduced CSF hypocretin-1 concentrations leading to greater alteration in sleep architecture. Furthermore, cytokine profiling in CSF revealed significant changes in interleukins 6 and 18 in NT1 patients, suggesting an active neuroinflammatory process possibly linked to ccf-mtDNA release, thus pointing to a specific inflammatory signature in NT1. These findings hint a potential mitochondrial dysfunction and neuroinflammation in NT1. Further studies are needed to elucidate the underlying mechanisms and how this may reflect on therapy.

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CiteScore
7.00
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