对患有罕见遗传性神经发育疾病的儿童进行行为睡眠干预:对 26 个病例总体疗效的回顾性分析

IF 1.5 Q3 EDUCATION, SPECIAL
Emma C. Woodford, Karyn G. France, Neville M. Blampied, Ursula Hanning, Catherine E. Swan, Laurie K. McLay
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引用次数: 0

摘要

目的:睡眠困难在患有罕见遗传神经发育疾病(RGNC)的儿童中非常普遍,如Angelman综合征和Smith-Magenis综合征。行为干预通常用于治疗儿童睡眠困难;然而,对RGNC患儿的研究有限。本研究评估了基于功能的行为睡眠干预对RGNC儿童的总体有效性和可接受性。方法对26名患有RGNC的儿童(18个月至19岁)进行了一系列的单例实验研究,这些儿童接受了行为睡眠干预。干预策略包括由父母实施的昼夜节律(例如,睡眠/觉醒重新安排),前事(例如,睡眠卫生)和/或后果(例如,积极强化睡眠有利行为和修正灭绝)修改。临床医生主要通过远程医疗方法为家长提供支持。总体结果采用改进的Brinley图和效应大小估计进行检验。年龄、性别和一系列心理变量对干预反应的影响也被检查。结果24/26名参与者的睡眠问题严重程度有所改善,13/16名参与者的睡眠问题严重程度在长期随访中保持改善。有趣的是,50%的儿童通过较少的限制性策略(例如,昼夜节律、先行策略和积极强化策略)实现了临床显著的变化,这表明消除程序可能被用作一系列干预措施中的最后一个选择。家长普遍认为干预是可以接受的。很少有证据表明,年龄、性别或心理变量对干预有任何不同的反应。结论基于功能的行为干预是治疗RGNC患儿睡眠障碍的有效方法。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions: A Retrospective Analysis of Overall Outcomes for 26 Cases

Objectives

Sleep difficulties are highly prevalent among children with rare genetic neurodevelopmental conditions (RGNC) such as Angelman and Smith-Magenis syndromes. Behavioral interventions are commonly used in the treatment of sleep difficulties in children; however, research is limited in children with RGNC. This study evaluated the overall effectiveness and acceptability of function-based behavioral sleep interventions for children with RGNC.

Methods

Data was collated from a series of experimental single-case research studies with 26 children (18 months to 19 years of age) with a range of RGNC, who received a behavioral sleep intervention. Intervention strategies included circadian (e.g., sleep/wake rescheduling), antecedent (e.g., sleep hygiene), and/or consequence (e.g., positive reinforcement of sleep-conducive behavior and modified extinction) modifications implemented by parents. Clinicians provided support for parents mostly via telehealth methods. Overall outcomes were examined using modified Brinley plots and effect size estimates. The effect of age, gender, and a range of psychological variables on intervention response was also examined.

Results

Improvements in sleep problem severity were observed for 24/26 participants, and gains were maintained at long-term follow-up for 13/16. Interestingly, 50% of the children achieved clinically significant change with less restrictive strategies (e.g., circadian, antecedent and positive reinforcement strategies), suggesting extinction procedures may be used as the last option in a sequence of interventions. Parents generally perceived interventions to be acceptable. There was minimal evidence of any differential response to intervention as a function of age, gender, or psychological variables.

Conclusions

Results suggest function-based behavioral interventions offer an effective and socially valid method for treating sleep disturbance in children with RGNC.

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来源期刊
Advances in Neurodevelopmental Disorders
Advances in Neurodevelopmental Disorders Social Sciences-Social Sciences (miscellaneous)
CiteScore
2.10
自引率
0.00%
发文量
72
期刊介绍: Advances in Neurodevelopmental Disorders publishes high-quality research in the broad area of neurodevelopmental disorders across the lifespan. Study participants may include individuals with:Intellectual and developmental disabilitiesGlobal developmental delayCommunication disordersLanguage disordersSpeech sound disordersChildhood-onset fluency disorders (e.g., stuttering)Social (e.g., pragmatic) communication disordersUnspecified communication disordersAutism spectrum disorder (ASD)Attention-deficit/hyperactivity disorder (ADHD), specified and unspecifiedSpecific learning disordersMotor disordersDevelopmental coordination disordersStereotypic movement disorderTic disorders, specified and unspecifiedOther neurodevelopmental disorders, specified and unspecifiedPapers may also include studies of participants with neurodegenerative disorders that lead to a decline in intellectual functioning, including Alzheimer’s disease, amyotrophic lateral sclerosis, Creutzfeldt-Jakob disease, vascular dementia, Lewy body dementia, frontotemporal dementia, corticobasal degeneration, Huntington’s disease, and progressive supranuclear palsy. The journal includes empirical, theoretical and review papers on a large variety of issues, populations, and domains, including but not limited to: diagnosis; incidence and prevalence; and educational, pharmacological, behavioral and cognitive behavioral, mindfulness, and psychosocial interventions across the life span. Animal models of basic research that inform the understanding and treatment of neurodevelopmental disorders are also welcomed. The journal is multidisciplinary and multi-theoretical, and encourages research from multiple specialties in the social sciences using quantitative and mixed-method research methodologies.
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