非随机重复经颅磁刺激治疗Rett综合征患儿有效性的临床试验

doi:10.11852/zgetbjzz2018-1810

中国儿童保健杂志 ›› 2019, Vol. 27 ›› Issue (7): 771-774.DOI: 10.11852/zgetbjzz2018-1810

非随机重复经颅磁刺激治疗Rett综合征患儿有效性的临床试验

王超^1,2, 李慧萍¹, 周秉睿¹, 李春阳¹, 张颖¹, 董萍¹, 李冬蕴¹, 鲁萍¹, 朱叶¹, 仇子龙³, 徐秀¹, 徐琼¹

1复旦大学附属儿科医院儿童保健科,上海 201102;
2吉林大学第一医院发育行为儿科,吉林长春130021;
3 中国科学院神经科学研究所神经科学国家重点实验室,上海 200031

收稿日期:2018-12-11 发布日期:2019-07-10 出版日期:2019-07-10
通讯作者: 徐琼,E-mail:xuqiong@fudan.edu.cn
作者简介:王超(1990-),女,黑龙江人,住院医师,硕士学位,主要研究方向为儿童发育行为相关疾病。
基金资助:
国家自然科学基金项目(81701496,81601327);上海市科学技术委员会(15411967900)

Non-randomized clinical trial of repetitive transcranial magnetic stimulation in the treatment of children with Rett syndrome

WANG Chao^1,2, LI Hui-ping¹, ZHOU Bing-rui¹, LI Chun-yang¹, ZHANG Ying¹, DONG Ping¹

1 Children′s Hospital of Fudan University,Department of Child Healthcare,Shanghai 201102,China;
2 The First Bethune Hospital of Jilin University,Department of Developmental and Behavioral Pediatrics,Jilin,Changchun 130021,China;
3 Institute of Neuroscience & State Key Laboratory of Neuroscience,Chinese Academy of Sciences,Shanghai 200031,China

Received:2018-12-11 Online:2019-07-10 Published:2019-07-10
Contact: XU Qiong,E-mail:xuqiong@fudan.edu.cn

摘要/Abstract

摘要： 目的分析重复经颅磁刺激(rTMS)治疗Rett综合征的有效性,为临床治疗提供依据。方法 2015年1月—2018年4月收集符合入组标准的RTT患儿17例,将其非随机分为TMS组(用rTMS治疗的干预组,7例)和对照组(10例),分别接受为期6个月的rTMS治疗和门诊常规随访。所有患儿均接受MECP2基因检测,并于入组时及6个月时分别接受Griffiths精神发育量表-中文版(GDS-C)、Peabody发育运动量表(PDMS)、儿童睡眠习惯问卷(CSHQ)、闪光视觉诱发电位(FVEP)及脑干听觉诱发电位(BAEP)的检查。方法 1)TMS组与对照组患儿在整体发育、运动能力方面均呈现明显的退行性变化过程,两组比较差异无统计学意义(P>0.05)。2)睡眠方面,TMS组及对照组患儿睡眠时间均较短,经过6个月的rTMS干预后,与对照组患儿相比,TMS组患儿平均睡眠时间延长,但差异无统计学意义[TMS组差值=(0.83± 0.97)h vs.对照组差值=(-0.40±1.20)h,P>0.05]。3)视听觉诱发电位检测提示,两组患儿治疗前后及组间比较亦无明显差异。4)TMS治疗过程中安全,未报告不良事件。结论 rTMS可能有延长RTT患儿睡眠时间、改善睡眠质量的作用。TMS治疗对于RTT患儿的智力和运动发育水平的倒退进程无改善作用,对RTT患儿的视觉及听觉电生理指标无明显影响。RTT患儿使用rTMS 过程中无严重不良反应。

关键词: 重复经颅磁刺激, Rett综合征, 睡眠时间, 精神运动发育, 视觉诱发电位, 听觉诱发电位

Abstract: Objectives To investigate the efficacy of repetitive transcranial magnetic stimulation(rTMS) in the treatment of Rett syndrome(RTT),so as to provide reference for clinical treatment. Methods Totally 17 children with Rett syndrome who met the criterion were enrolled in this study from January 2015 to April 2018,and were non-randomly divided into the TMS group(n=7) and control group(n=10). The TMS group was given rTMS for 6 months,while the control group received routine follow-up. All children were tested for MECP2 gene and were assessed by Griffiths Mental Development Scale-Chinese Version(GDS-C),Peabody Development Motor Scales(PDMS),Children′s Sleep Habits Questionnaire(CSHQ),flash visual evoked potential(FVEP) and brainstem auditory evoked potential(BAEP) at admission and 6 months respectively. Results 1) The overall development and motor ability of the children in TMS group and control group showed obviously degenerative changes,but the differences between the two groups were not significant(P＞0.05). 2) The sleep duration of children in TMS group and control group was shorter. After 6 months of rTMS intervention in TMS group,the average sleep duration of children in TMS group was longer than that of control group,but there was no significant difference between the two groups[ΔTMS group:(0.83±0.97) h vs. Δ control group:(-0.40±1.20) h,P＞0.05]. 3) There were no significant changes on the results of FVEP and BAEP before and after treatment,so was the difference between the two groups(P＞0.05). 4) rTMS was safe during treatment and no adverse events were reported. Conclusions Repeated transcranial magnetic stimulation may prolong the sleep duration and improve the quality of sleep in RTT children. Moreover,rTMS treatment has no significant effect on the progression of mental and motor developmental levels in RTT children,as well as the parameters of FVEP and BAEP. Meanwhile,there are no serious adverse reactions during the use of rTMS

Key words: repetitive transcranial magnetic stimulation, Rett syndrome, sleep duration, psychomotor development, visual evoked potential, auditory evoked potential

中图分类号:

R749.94

王超, 李慧萍, 周秉睿, 李春阳, 张颖, 董萍, 李冬蕴, 鲁萍, 朱叶, 仇子龙, 徐秀, 徐琼. 非随机重复经颅磁刺激治疗Rett综合征患儿有效性的临床试验[J]. 中国儿童保健杂志, 2019, 27(7): 771-774.

WANG Chao, LI Hui-ping, ZHOU Bing-rui, LI Chun-yang, ZHANG Ying, DONG Ping. Non-randomized clinical trial of repetitive transcranial magnetic stimulation in the treatment of children with Rett syndrome[J]. journal1, 2019, 27(7): 771-774.

参考文献

[1] Neul JL,Kaufmann WE,Glaze DG,et al.Rett syndrome:revised diagnostic criteria and nomenclature[J].Ann Neurol,2010,68(6):944-950.
[2] Liyanage VR,Rastegar M.Rett syndrome and MeCP2[J].Neuromol Med,2014,16(2):231-264.
[3] Tillotson R,Selfridge J,Koerner MV,et al.Radically truncated MeCP2 rescues Rett syndrome-like neurological defects[J].Nature,2017,550(7676):398-401.
[4] Carrette LLG,BlumR,MaW,et al.Tsix-Mecp2 female mous e model for Rett syndrome reveals that low-level MECP2 expression extends life and improves neuromotor function[J].P National Acad Sci USA,2018,115(32):8185-8190.
[5] Balakrishnan S,Mironov SL.Rescue of hyperexcitability in hippocampal CA1 neurons from Mecp2(-/y)mouse through surface potential neutralization[J].PLoS One,2018,13(4):e0195094.
[6] Xiao L,Correll CU,Feng L,et al.Rhythmic low-field magnetic stimulation may improve depression by increasing brain-derived neurotrophic factor[J].CNS Spectr,2019,24(3):313-321.
[7] Landeros-Weisenberger A,Mantovani A,Motlagh MG,et al.Randomized sham controlled double-blind trial of repetitive transcranial magnetic stimulation for adults with severe tourette syndrome[J].Brain Stimul,2015,8(3):574-581.
[8] Lee YJ,Koo BH,Seo WS,et al.Repetitive transcranial magnetic stimulation of the supplementary motor area in treatment-resistant obsessive-compulsive disorder:an open-label pilot study[J].J Clin Neurosci,2017,44:264-268.
[9] Barahona-Correa JB,Velosa A,Chainho A,et al.Repetitive transcranial magnetic stimulation for treatment of autism spectrum disorder:a systematic review and meta Analysis[J].Front Integr Neurosci,2018,12:27.
[10] Eeíková A,Jech R, ejka V,et al.Benefits of pallidal stimulation in dystonia are linked to cerebellar volume and cortical inhibition[J].Sci Rep,2018,8(1):17218.
[11] Chou YH,Hickey PT,Sundman M,et al.Effects of repetitive transcranial magnetic stimulation on motor symptoms in Parkinson disease:a systematic review and meta-analysis[J].JAMA Neurol,2015,72(4):432-440.
[12] Yang C,Guo Z,Peng H,et al.Repetitive transcranial magnetic stimulation therapy for motor recovery in Parkinson′s disease:a Meta-analysis[J].Brain Behav,2018,8(11):e01132.
[13] Iimori T,Nakajima S,Miyazaki T,et al.Effectiveness of the prefrontal repetitive transcranial magnetic stimulation on cognitive profiles in depression,schizophrenia,and Alzheimer′s disease:a systematic review[J].Prog Neuro-Psychoph,2019,88:31-40.
[14] Zhang Y,Mao RR,Chen ZF,et al.Deep-brain magnetic stimulation promotes adult hippocampal neurogenesis and alleviates stress-related behaviors in mouse models for neuropsychiatric disorders[J].Mol Brain,2014,7:11.
[15] Hermiller MS,Vanhaerents S,Raij T,et al.Frequency-specific noninvasive modulation of memory retrieval and its relationship with hippocampal network connectivity[J].Hippocampus,2018.doi:10.1002/hipo.23054.
[16] Owen,JA,Spirito A,McGuinn M.The Children′s Sleep Habits Questionnaire(CSHQ):psyshome tric propen ties of a survey instrument for schoolaged children[J].Sleep,2000,23(8):1043-1051.
[17] 陈文娟,李锋,李生慧,等.常用儿童睡眠时间评估方法的比较研究[J].中华儿科杂志,2012,50(4):5.
[18] Liao W.Psychomotor dysfunction in Rett syndrome:insights into the neurochemical and circuit roots[J].Dev Neurobiol,2019,79(1):51-59.
[19] Wu Y,Cui N,Xing H,et al.Mecp2 disruption in rats causes reshaping in firing activity and patterns of brainstem respiratory neurons[J].Neuroscience,2019,397:107-115.
[20] Amaddeo A,Desanctis L,Arroyo JO,et al.Polysomnograp- hic findings in Rett syndrome[J].Eur J Paediatr Neurol,2019,23(1):214-221.
[21] Cappuccio G,Bernardo P,Raiano E,et al.Pain and sleep disturbances in Rett syndrome and other neurodevelopmental disorders[J].Acta Paediatr,2019,108(1):171-172.
[22] Nishida M,Kikuchi S,Nisijima K,et al.Actigraphy in patients with major depressive disorder undergoing repetitive transcranial magnetic stimulation:an open label pilot study[J].J ECT,2017,33(1):36-42.
[23] LeBlanc JJ,DeGregorio G,Centofante E,et al.Visual evok- ed potentials detect cortical processing deficits in Rett syndrome[J].Ann Neurol,2015,78(5):775-86.
[24] Foxe JJ,Burke KM,Andrade GN,et al.Automatic cortical representation of auditory pitch changes in Rett syndrome[J].J Neurodev Disord,2016,8(1):34.
[25] Pereira LS,Muller VT,da Mota Gomes M,et al.Safety of repetitive transcranial magnetic stimulation in patients with epilepsy:a systematic review[J].Epilepsy Behav,2016,57:167-176

非随机重复经颅磁刺激治疗Rett综合征患儿有效性的临床试验

Non-randomized clinical trial of repetitive transcranial magnetic stimulation in the treatment of children with Rett syndrome

PDF

可视化

摘要/Abstract

引用本文

使用本文

参考文献

相关文章 15

编辑推荐

Metrics

[1]	王惠萍, 王莉, 高琼, 白铂亮, 马颖君. 极低出生体重坏死性小肠结肠炎患儿体格及神经发育的研究[J]. 中国儿童保健杂志, 2023, 31(1): 96-100.
[2]	龙姿冲, 陈仪婷, 赵安达, 黄利利, 夏缘青, 吕佳骏, 李生慧. 就寝-晨起时间及睡眠节律模式与青少年过敏性鼻炎的相关性研究[J]. 中国儿童保健杂志, 2022, 30(3): 254-258.
[3]	黄国书, 刘莹, 黄炳谕, 梁藐英. 经颅磁刺激联合功能电刺激对全面发育迟缓患儿的疗效分析[J]. 中国儿童保健杂志, 2022, 30(11): 1169-1172.
[4]	谢巧玲. MSTO1基因新变异所致精神运动发育迟缓患儿的基因分析[J]. 中国儿童保健杂志, 2021, 29(9): 1038-1040.
[5]	宁书尧, 陈文雄, 杨思渊, 李嘉铃, 梁华冰, 胡优秀. 华南地区788例孤独症谱系障碍儿童脑干听觉诱发电位的研究[J]. 中国儿童保健杂志, 2021, 29(7): 775-778.
[6]	许鑫悦, 刘洋. Rett综合征伴真性性早熟1例病例报道[J]. 中国儿童保健杂志, 2021, 29(2): 230-232.
[7]	张学荣, 陈天娇, 马军. 学龄儿童睡眠时间与超重肥胖的相关性[J]. 中国儿童保健杂志, 2021, 29(11): 1176-1180.
[8]	贠国俊, 王景刚, 李瑞豪, 佟南, 曹建国, 郭莹莹. 重复经颅磁联合体外冲击波对偏瘫患儿下肢功能的临床研究[J]. 中国儿童保健杂志, 2021, 29(11): 1265-1268.
[9]	黄怡颖, 王文强, 吴为阁, 李玉妹. 注意缺陷多动障碍和孤独症谱系障碍患者脑干听觉诱发电位比较[J]. 中国儿童保健杂志, 2020, 28(7): 740-743.
[10]	温勃, 董彦会, 杨招庚, 王西婕, 李艳辉, 董彬, 邹志勇, 马军. 中国9~14岁青春期女生睡眠时间与月经初潮的关系[J]. 中国儿童保健杂志, 2019, 27(7): 701-705.
[11]	王小燕, 张萍, 汪鸿, 赵职卫. 6月持续脑干诱发电位异常对2岁婴幼儿神经心理发育的预测研究[J]. 中国儿童保健杂志, 2019, 27(4): 433-435.
[12]	刘黎礼,铁晓玲. 精细运动发育落后幼儿视觉运动整合与视诱发电位关系探讨[J]. 中国儿童保健杂志, 2019, 27(3): 308-310.
[13]	张萍,王小燕,汪鸿,赵职卫. 正常328名1~6月龄婴儿脑干听觉诱发电位结果分析[J]. 中国儿童保健杂志, 2019, 27(3): 311-313.
[14]	柳秋菊. 新生儿期高胆红素血症患儿3月龄神经行为评估结果分析[J]. 中国儿童保健杂志, 2019, 27(2): 183-185.
[15]	林力孜, 高爱钰 ,王迪,承钰, 程兰 ,李钦 ,王海俊. 小学生睡眠时间和视屏时间与儿童肥胖的关联研究[J]. 中国儿童保健杂志, 2018, 26(9): 948-951.