Motor and sensory neurophysiology in relation to [18F]FDG PET imaging in children with dystonia.

IF 4.5 Q1 CLINICAL NEUROLOGY
Brain communications Pub Date : 2025-06-18 eCollection Date: 2025-01-01 DOI:10.1093/braincomms/fcaf151
Stavros Tsagkaris, Verity McClelland, Doreen Fialho, Daniel E Lumsden, Margaret Kaminska, Eric Guedj, Alexander Hammers, Jean-Pierre Lin
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引用次数: 0

Abstract

The network model of dystonia reconciles many of the neuroanatomical and electrophysiological abnormalities identified as potential pathophysiological factors. Recent [18F]fluorodeoxyglucose-PET brain imaging findings support this concept, revealing distinct patterns of abnormal brain metabolism in specific dystonia aetiologies. However, it is unclear how changes in specific neural pathways alter brain glucose metabolism. This observational study investigates patterns of brain metabolism using [18F]fluorodeoxyglucose-PET imaging in children with dystonia, awake-resting during the uptake period, in relation to measures of brain function using standard neurophysiological tests of motor and sensory pathway integrity. Central motor conduction times, somatosensory evoked potentials and [18F] fluorodeoxyglucose-PET scans were obtained in children with dystonia or dystonic-dyskinetic cerebral palsy undergoing standard clinical assessment for bilateral pallidal deep brain stimulation between 2007 and 2018 in Evelina London Children's Hospital. Data from 109 children aged 2.8-18.8 years were analysed retrospectively. Patients were divided into groups based on their neurophysiology results as follows: both tests normal (NN; 67), both abnormal (AA; 11), normal central motor conduction times/abnormal somatosensory evoked potentials (NA; 20), abnormal central motor conduction times/normal somatosensory evoked potentials (AN; 11). Groups were compared with a control group comprising [18F]fluorodeoxyglucose-PET scans from 39 healthy adults using Statistical Parametric Mapping 12 with age and groupwise global means as covariates. Taking into account groupwise global uptake, all four groups shared relative hypermetabolism in parietal areas, postcentral and precentral gyri. In addition, mild peri-insular hypometabolism was seen in the NN group. The NA group showed marked regional hypometabolism bilaterally in the thalami, globi pallidi, putamina, heads of the caudate nuclei and areas of peri-sylvian cortex. The AN group had hypometabolism in the thalami and posterior globi pallidi, the posterior putamina and areas of peri-sylvian cortex. The AA group also exhibited hypometabolism in the medial thalami and some areas of frontal and peri-insular cortex. Across the whole cohort, abnormal somatosensory evoked potentials were strongly associated with thalamic hypometabolism, with no marked differences for abnormal central motor conduction times. Brain metabolism patterns in dystonia relate to neurophysiological abnormalities in our study. Relative parietal hypermetabolism is more common than recognized previously, while thalamic hypometabolism is prominent in those with abnormal sensory pathway function. The findings support the network model of dystonia and emphasize the importance of multi-modal assessment in providing detailed phenotyping, which could inform individualized management strategies.

运动和感觉神经生理学与肌张力障碍儿童FDG PET成像的关系[18F]。
肌张力障碍的网络模型协调了许多神经解剖和电生理异常,这些异常被认为是潜在的病理生理因素。最近的[18F]氟脱氧葡萄糖- pet脑成像结果支持这一概念,揭示了特定肌张力障碍病因中不同的脑代谢异常模式。然而,目前尚不清楚特定神经通路的变化如何改变脑葡萄糖代谢。本观察性研究使用[18F]氟脱氧葡萄糖- pet成像研究摄取期间肌张力障碍患儿的脑代谢模式,并使用运动和感觉通路完整性的标准神经生理学测试测量脑功能。对2007年至2018年在Evelina伦敦儿童医院接受双侧侧深部脑刺激标准临床评估的肌张力障碍或肌张力-运动障碍脑瘫患儿进行中枢运动传导次数、体感诱发电位和[18F]氟氧葡萄糖- pet扫描。回顾性分析了109名2.8-18.8岁儿童的数据。根据神经生理学结果将患者分为两组:两组均正常(NN;67),均为异常(AA;11)、正常中枢运动传导次数/异常体感诱发电位(NA;20),异常中枢运动传导次数/正常体感诱发电位(AN);11)。各组与对照组进行比较,对照组由39名健康成人的[18F]氟脱氧葡萄糖- pet扫描组成,使用统计参数映射12,以年龄和分组全球均值为协变量。考虑到各组整体摄取,所有四组在顶叶区,中央后和中央前回都有相对的高代谢。此外,神经网络组出现轻度岛周低代谢。NA组双侧丘脑、苍白球、壳层、尾状核头部和外侧皮层区域均出现明显的区域性低代谢。AN组丘脑和后苍白球、后壳层和周围皮层区域代谢降低。AA组在丘脑内侧、额叶和岛叶周围皮层的部分区域也表现出代谢低下。在整个队列中,异常的体感诱发电位与丘脑低代谢密切相关,而异常的中枢运动传导时间没有显著差异。在我们的研究中,肌张力障碍的脑代谢模式与神经生理异常有关。相对顶叶高代谢比以前认识到的更常见,而丘脑低代谢在感觉通路功能异常的患者中更为突出。这些发现支持肌张力障碍的网络模型,并强调了多模式评估在提供详细表型方面的重要性,这可以为个性化的管理策略提供信息。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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