Electroencephalographic patterns during sleep in children with chromosome 15q11.2-13.1 duplications (Dup15q)

Electroencephalographic patterns during sleep in children with chromosome 15q11.2-13.1 duplications (Dup15q)
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DOI:
10.1016/j.yebeh.2016.02.010
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发表时间:
2016-04-01
影响因子:
2.6
通讯作者:
Thibert, Ronald
Thibert, Ronald
中科院分区:
医学3区
文献类型:
--
作者:
Arkilo, Dimitrios;Devinsky, Orrin;Thibert, Ronald

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我们的目的是明确因15q11-q13(Dup1 5q)拷贝数增加而导致的神经发育障碍儿童睡眠期间的脑电特征。我们回顾了42例Dupl5q儿童(平均年龄:8岁,32例)的连续脑电记录,收集的数据包括背景活动、发作间期癫痫样放电、睡眠组织和发作性活动。有三种模式:模式1:14名儿童(33%)出现阿尔法-德尔塔睡眠,与任何临床变化无关。模式2:15名儿童(35%)出现睡眠中的癫痫电状态,均被诊断为难治性癫痫。15名儿童中有13名有临床癫痫发作。模式3:15例(35%)儿童在非REM睡眠中出现以双额叶高波幅为主的阵发性快速活动(12~15 Hz)。所有15名儿童都患有难治性癫痫。这是首次报道Dup15q儿童睡眠期间的脑电模式,Dup15q报告阿尔法增量节律、CSWS和高幅度快频率。有自主神经障碍和/或情绪障碍的儿童和发育退化的儿童中描述了阿尔法-德尔塔节律。这些发现对我们队列中的儿童的认知功能和癫痫的意义需要通过后续研究来确定。(C)2016 Elsevier Inc.保留所有权利。
Our objective was to define the EEG features during sleep of children with neurodevelopmental disorders due to copy number gains of 15q11-q13 (Dupl 5q). We retrospectively reviewed continuous EEG recordings of 42 children with Dupl5q (mean age: eight years, 32 with idicl5), and data collected included background activity, interictal epileptiform discharges, sleep organization, and ictal activity. Three patterns were recognized:Pattern 1: Alpha-delta sleep was noted in 14 children (33%), not associated with any clinical changes. Pattern 2: Electrical status epilepticus in sleep was noted in 15 children (35%), all diagnosed with treatment resistant epilepsy. Thirteen of the 15 children had clinical seizures. Pattern 3: Frequent bursts of high amplitude bifrontal predominant, paroxysmal fast activity (12-15 Hz) during non-REM sleep was noted in 15 children (35%). All 15 children had treatment-resistant epilepsy.This is the first report of electroencephalographic patterns during sleep of children with Dup15q reporting alpha delta rhythms, CSWS, and high amplitude fast frequencies. Alpha-delta rhythms are described in children with dysautonomia and/or mood disorders and CSWS in children with developmental regression. The significance of these findings in cognitive function and epilepsy for the children in our cohort needs to be determined with follow-up studies. (C) 2016 Elsevier Inc. All rights reserved.