Aberrant oscillatory activity in neurofibromatosis type 1: an EEG study of resting state and working memory.

IF 4.1 2区 医学 Q1 CLINICAL NEUROLOGY Journal of Neurodevelopmental Disorders Pub Date : 2023-08-22 DOI:10.1186/s11689-023-09492-y
Samantha J Booth, Shruti Garg, Laura J E Brown, Jonathan Green, Gorana Pobric, Jason R Taylor
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Abstract

Background: Neurofibromatosis type 1 (NF1) is a genetic neurodevelopmental disorder commonly associated with impaired cognitive function. Despite the well-explored functional roles of neural oscillations in neurotypical populations, only a limited number of studies have investigated oscillatory activity in the NF1 population.

Methods: We compared oscillatory spectral power and theta phase coherence in a paediatric sample with NF1 (N = 16; mean age: 13.03 years; female: n = 7) to an age/sex-matched typically developing control group (N = 16; mean age: 13.34 years; female: n = 7) using electroencephalography measured during rest and during working memory task performance.

Results: Relative to typically developing children, the NF1 group displayed higher resting state slow wave power and a lower peak alpha frequency. Moreover, higher theta power and frontoparietal theta phase coherence were observed in the NF1 group during working memory task performance, but these differences disappeared when controlling for baseline (resting state) activity.

Conclusions: Overall, results suggest that NF1 is characterised by aberrant resting state oscillatory activity that may contribute towards the cognitive impairments experienced in this population.

Trial registration: ClinicalTrials.gov, NCT03310996 (first posted: October 16, 2017).

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1型神经纤维瘤病的异常振荡活动:静息状态和工作记忆的脑电图研究。
背景:1型神经纤维瘤病(NF1)是一种遗传性神经发育障碍,通常与认知功能受损有关。尽管神经振荡在神经正常人群中的功能作用得到了很好的探索,但只有有限数量的研究调查了NF1人群的振荡活动。方法:我们比较了小儿NF1 (N = 16;平均年龄13.03岁;女性:n = 7)到年龄/性别匹配的典型发育对照组(n = 16);平均年龄13.34岁;女性:n = 7)在休息和工作记忆任务执行期间使用脑电图测量。结果:与正常发育儿童相比,NF1组静息状态慢波功率较高,α峰频率较低。此外,NF1组在工作记忆任务执行过程中观察到更高的θ波功率和额顶叶θ波相相干性,但在控制基线(静息状态)活动时,这些差异消失。结论:总的来说,结果表明NF1的特征是异常的静息状态振荡活动,这可能导致该人群经历的认知障碍。试验注册:ClinicalTrials.gov, NCT03310996(首次发布时间:2017年10月16日)。
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来源期刊
CiteScore
7.60
自引率
4.10%
发文量
58
审稿时长
>12 weeks
期刊介绍: Journal of Neurodevelopmental Disorders is an open access journal that integrates current, cutting-edge research across a number of disciplines, including neurobiology, genetics, cognitive neuroscience, psychiatry and psychology. The journal’s primary focus is on the pathogenesis of neurodevelopmental disorders including autism, fragile X syndrome, tuberous sclerosis, Turner Syndrome, 22q Deletion Syndrome, Prader-Willi and Angelman Syndrome, Williams syndrome, lysosomal storage diseases, dyslexia, specific language impairment and fetal alcohol syndrome. With the discovery of specific genes underlying neurodevelopmental syndromes, the emergence of powerful tools for studying neural circuitry, and the development of new approaches for exploring molecular mechanisms, interdisciplinary research on the pathogenesis of neurodevelopmental disorders is now increasingly common. Journal of Neurodevelopmental Disorders provides a unique venue for researchers interested in comparing and contrasting mechanisms and characteristics related to the pathogenesis of the full range of neurodevelopmental disorders, sharpening our understanding of the etiology and relevant phenotypes of each condition.
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