Angelman综合征的定量脑电图分析:评估治疗方法的候选方法。

IF 1.6 4区 医学 Q3 CLINICAL NEUROLOGY Clinical EEG and Neuroscience Pub Date : 2023-03-01 DOI:10.1177/1550059420973095
Luis A Martinez, Heather A Born, Sarah Harris, Angelique Regnier-Golanov, Joseph C Grieco, Edwin J Weeber, Anne E Anderson
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引用次数: 10

摘要

这些研究的目的是使用定量(q)脑电图技术对Angelman综合征(AS)儿童的数据进行谱功率分析,并评估其作为潜在的生物标志物和定量方法来评估治疗方法。虽然特征模式在目视检查中很明显,但使用qEEG技术有可能提供治疗效果的定量证据。我们首先评估了从AS儿童收集的基线脑电图记录的频谱功率,并将其与年龄匹配的神经典型对照进行比较,这证实了先前报道的发现,即AS儿童的δ功率升高导致总功率增加。然后,我们回顾性分析了在评估米诺环素(3mg /kg/d)的安全性和耐受性的临床试验中收集的数据,以比较治疗结束时AS儿童(4-12岁)的脑电图活动记录和洗净后的脑电图频谱功率和癫痫样事件。在基线和米诺环素治疗期间,AS受试者表现出增加的δ功率;然而,在米诺环素治疗后,AS受试者的脑电图频谱功率和癫痫样活动显著降低。我们的研究结果支持qEEG分析在评估AS中的应用,并表明该技术可能有助于评估AS的治疗效果。因此,脑电图功率正常化可能成为筛选治疗方法以衡量整体疗效的重要指标。随着治疗方法从临床前研究过渡到临床研究,建立能够定量评估具有独特脑电图模式的As和神经系统疾病的推定治疗方法的结果测量是至关重要的。
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Quantitative EEG Analysis in Angelman Syndrome: Candidate Method for Assessing Therapeutics.

The goal of these studies was to use quantitative (q)EEG techniques on data from children with Angelman syndrome (AS) using spectral power analysis, and to evaluate this as a potential biomarker and quantitative method to evaluate therapeutics. Although characteristic patterns are evident in visual inspection, using qEEG techniques has the potential to provide quantitative evidence of treatment efficacy. We first assessed spectral power from baseline EEG recordings collected from children with AS compared to age-matched neurotypical controls, which corroborated the previously reported finding of increased total power driven by elevated delta power in children with AS. We then retrospectively analyzed data collected during a clinical trial evaluating the safety and tolerability of minocycline (3 mg/kg/d) to compare pretreatment recordings from children with AS (4-12 years of age) to EEG activity at the end of treatment and following washout for EEG spectral power and epileptiform events. At baseline and during minocycline treatment, the AS subjects demonstrated increased delta power; however, following washout from minocycline treatment the AS subjects had significantly reduced EEG spectral power and epileptiform activity. Our findings support the use of qEEG analysis in evaluating AS and suggest that this technique may be useful to evaluate therapeutic efficacy in AS. Normalizing EEG power in AS therefore may become an important metric in screening therapeutics to gauge overall efficacy. As therapeutics transition from preclinical to clinical studies, it is vital to establish outcome measures that can quantitatively evaluate putative treatments for AS and neurological disorders with distinctive EEG patterns.

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来源期刊
Clinical EEG and Neuroscience
Clinical EEG and Neuroscience 医学-临床神经学
CiteScore
5.20
自引率
5.00%
发文量
66
审稿时长
>12 weeks
期刊介绍: Clinical EEG and Neuroscience conveys clinically relevant research and development in electroencephalography and neuroscience. Original articles on any aspect of clinical neurophysiology or related work in allied fields are invited for publication.
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