Emma C. Woodford, Karyn G. France, Neville M. Blampied, Ursula Hanning, Catherine E. Swan, Laurie K. McLay
{"title":"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","authors":"Emma C. Woodford, Karyn G. France, Neville M. Blampied, Ursula Hanning, Catherine E. Swan, Laurie K. McLay","doi":"10.1007/s41252-024-00403-3","DOIUrl":null,"url":null,"abstract":"<div><h3>Objectives</h3><p>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.</p><h3>Methods</h3><p>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.</p><h3>Results</h3><p>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.</p><h3>Conclusions</h3><p>Results suggest function-based behavioral interventions offer an effective and socially valid method for treating sleep disturbance in children with RGNC.</p></div>","PeriodicalId":36163,"journal":{"name":"Advances in Neurodevelopmental Disorders","volume":"9 2","pages":"265 - 280"},"PeriodicalIF":1.5000,"publicationDate":"2024-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://link.springer.com/content/pdf/10.1007/s41252-024-00403-3.pdf","citationCount":"0","resultStr":"{\"title\":\"Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions: A Retrospective Analysis of Overall Outcomes for 26 Cases\",\"authors\":\"Emma C. Woodford, Karyn G. France, Neville M. Blampied, Ursula Hanning, Catherine E. Swan, Laurie K. McLay\",\"doi\":\"10.1007/s41252-024-00403-3\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Objectives</h3><p>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.</p><h3>Methods</h3><p>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.</p><h3>Results</h3><p>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.</p><h3>Conclusions</h3><p>Results suggest function-based behavioral interventions offer an effective and socially valid method for treating sleep disturbance in children with RGNC.</p></div>\",\"PeriodicalId\":36163,\"journal\":{\"name\":\"Advances in Neurodevelopmental Disorders\",\"volume\":\"9 2\",\"pages\":\"265 - 280\"},\"PeriodicalIF\":1.5000,\"publicationDate\":\"2024-05-07\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://link.springer.com/content/pdf/10.1007/s41252-024-00403-3.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Advances in Neurodevelopmental Disorders\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://link.springer.com/article/10.1007/s41252-024-00403-3\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"EDUCATION, SPECIAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Advances in Neurodevelopmental Disorders","FirstCategoryId":"1085","ListUrlMain":"https://link.springer.com/article/10.1007/s41252-024-00403-3","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"EDUCATION, SPECIAL","Score":null,"Total":0}
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.
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.