PDF(Pubmed)
Abstract：
UNASSIGNED: Tuberous Sclerosis Complex (TSC) manifests behaviorally with features of autism, epilepsy, and intellectual disability. Resting state electroencephalography (EEG) offers a window into neural oscillatory activity and may serve as an intermediate biomarker between gene expression and behavioral manifestations. Such a biomarker could be useful in clinical trials as an endpoint or predictor of treatment response. However, seizures and antiepileptic medications also affect resting neural oscillatory activity and could undermine the utility of resting state EEG features as biomarkers in neurodevelopmental disorders such as TSC.
UNASSIGNED: This paper compares resting state EEG features in a cross-sectional cohort of young children with TSC (n=49, ages 12-37 months) to 49 age- and sex-matched typically developing controls. Within children with TSC, associations were examined between resting state EEG features, seizure severity composite score, and use of GABA agonists.
UNASSIGNED: Compared to matched typically developing controls, children with TSC showed significantly greater alpha and beta power in permutation cluster analyses iterated across a broad frequency range (2-50Hz). Children with TSC also showed significantly greater aperiodic offset after power spectra were parameterized using SpecParam into aperiodic and periodic components. Within children with TSC, greater seizure severity was significantly related to increased periodic peak beta power. Use of GABA agonists was also independently and significantly associated with increased periodic peak beta power; the interaction between seizure severity and GABA agonist use had no significant effect on peak beta power.
UNASSIGNED: The elevated peak beta power observed in children with TSC compared to matched typically developing controls may be driven by both seizures and GABA agonist use. It is recommended to collect seizure and mediation data alongside EEG data for clinical trials. These results highlight the challenge of using resting state EEG features as biomarkers in trials with neurodevelopmental disabilities when epilepsy and anti-epileptic medication are common.
UNASSIGNED: This paper compares resting state EEG features in a cross-sectional cohort of young children with TSC (n=49, ages 12-37 months) to 49 age- and sex-matched typically developing controls. Within children with TSC, associations were examined between resting state EEG features, seizure severity composite score, and use of GABA agonists.
UNASSIGNED: Compared to matched typically developing controls, children with TSC showed significantly greater alpha and beta power in permutation cluster analyses iterated across a broad frequency range (2-50Hz). Children with TSC also showed significantly greater aperiodic offset after power spectra were parameterized using SpecParam into aperiodic and periodic components. Within children with TSC, greater seizure severity was significantly related to increased periodic peak beta power. Use of GABA agonists was also independently and significantly associated with increased periodic peak beta power; the interaction between seizure severity and GABA agonist use had no significant effect on peak beta power.
UNASSIGNED: The elevated peak beta power observed in children with TSC compared to matched typically developing controls may be driven by both seizures and GABA agonist use. It is recommended to collect seizure and mediation data alongside EEG data for clinical trials. These results highlight the challenge of using resting state EEG features as biomarkers in trials with neurodevelopmental disabilities when epilepsy and anti-epileptic medication are common.
摘要：
背景：结节性硬化症（TSC）表现为自闭症的行为特征，癫痫,智力残疾。静息状态脑电图（EEG）提供了进入神经振荡活动的窗口，并且可以作为基因表达和行为表现之间的中间生物标志物。这样的生物标志物可以在临床试验中用作治疗反应的终点或预测因子。然而,癫痫发作和抗癫痫药物也会影响静息神经振荡活动，并可能破坏静息状态EEG特征作为神经发育障碍如TSC的生物标志物的效用。
方法：本文比较了一个由TSC（n=49，年龄12-37个月）与49个年龄和性别匹配的典型对照儿童组成的横断面队列中的静息状态EEG特征。在患有TSC的儿童中，研究了静息状态脑电图特征之间的关联，癫痫发作严重程度综合评分，以及GABA激动剂的使用。
结果：与匹配的典型开发对照相比，在宽频率范围(2-50Hz)内迭代的排列聚类分析中,TSC患儿的α和β功率显著增加.使用SpecParam将功率谱参数化为非周期性和周期性分量后，患有TSC的儿童也显示出明显更大的非周期性偏移。在患有TSC的儿童中，更大的癫痫发作严重程度与周期性峰值β功率增加显著相关.GABA激动剂的使用也与周期性峰值β功率增加独立且显着相关；癫痫发作严重程度和GABA激动剂使用之间的相互作用对峰值β功率没有显着影响。
结论：与匹配的典型发育对照相比，在TSC儿童中观察到的峰值β功率升高可能是由癫痫发作和GABA激动剂使用引起的。建议收集癫痫发作和调解数据以及脑电图数据进行临床试验。这些结果突出了在癫痫和抗癫痫药物常见的情况下，使用静息状态EEG特征作为神经发育障碍试验中的生物标志物的挑战。
方法：本文比较了一个由TSC（n=49，年龄12-37个月）与49个年龄和性别匹配的典型对照儿童组成的横断面队列中的静息状态EEG特征。在患有TSC的儿童中，研究了静息状态脑电图特征之间的关联，癫痫发作严重程度综合评分，以及GABA激动剂的使用。
结果：与匹配的典型开发对照相比，在宽频率范围(2-50Hz)内迭代的排列聚类分析中,TSC患儿的α和β功率显著增加.使用SpecParam将功率谱参数化为非周期性和周期性分量后，患有TSC的儿童也显示出明显更大的非周期性偏移。在患有TSC的儿童中，更大的癫痫发作严重程度与周期性峰值β功率增加显著相关.GABA激动剂的使用也与周期性峰值β功率增加独立且显着相关；癫痫发作严重程度和GABA激动剂使用之间的相互作用对峰值β功率没有显着影响。
结论：与匹配的典型发育对照相比，在TSC儿童中观察到的峰值β功率升高可能是由癫痫发作和GABA激动剂使用引起的。建议收集癫痫发作和调解数据以及脑电图数据进行临床试验。这些结果突出了在癫痫和抗癫痫药物常见的情况下，使用静息状态EEG特征作为神经发育障碍试验中的生物标志物的挑战。
