Promoting Prosocial Behavior in Syndromic Intellectual and Developmental Disabilities

促进综合症性智力和发育障碍的亲社会行为

• 批准号: 10363147
• 负责人: LATHA Valluripalli SOORYA
• 金额: $ 81.34万
• 依托单位: RUSH UNIVERSITY MEDICAL CENTER
• 依托单位国家: 美国
• 财政年份: 2022
• 资助国家: 美国
• 起止时间: 2022-07-15 至 2025-06-30
• 项目状态: 未结题
• 来源: https://reporter.nih.gov/project-details/10363147
• 关键词: Address; Adult; Affect; Age; Angelman Syndrome; Behavior; Behavior Therapy; Behavioral; Blinded; COVID-19 impact; COVID-19 pandemic; Caregiver support; Caregivers; Caring; Child; Child Behavior; Child Rearing; Clinical; Clinical effectiveness; Cognitive; Communication; Communities; Complex; Data; Effectiveness; Environment; Equipment and supply inventories; Family; Genetic; Goals; Health Services; Health Services Accessibility; Healthcare; Home; Hybrids; Impairment; Individual; Intellectual functioning disability; Intervention; Interview; Life; Longevity; Measures; Medical; Mental Health; Methods; Modeling; Modification; Motor; Natural History; Neurobiology; Outcome; Parents; Participant; Patients; Phelan-McDermid syndrome; Phenotype; Population; Population Genetics; Provider; Publishing; Questionnaires; Randomized; Randomized Controlled Trials; Reporting; Research; Resources; Services; Standardization; Subgroup; Surveys; Syndrome; Testing; Time; Tuberous Sclerosis; Vulnerable Populations; Work; applied behavior analysis; base; behavioral health; behavioral phenotyping; behavioral response; care systems; communication behavior; design; effective intervention; effectiveness implementation study; effectiveness-implementation RCT; evidence base; experience; functional disability; health inequalities; implementation science; impression; improved; individuals with autism spectrum disorder; innovation; intervention delivery; pandemic disease; person centered; programs; psychosocial; reinforced behavior; screening; severe intellectual disability; social; standardize measure; telehealth; treatment as usual; virtual

Project Summary/Abstract: The enormous impact of COVID-19 service disruptions on individuals with intellectual and developmental disabilities (IDDs) has highlighted the critically urgent need to increase access to mental and behavioral health services. Within IDD populations, genetic syndromes associated with IDDs (“syndromic IDDs”) represent a particularly vulnerable subgroup. Many patients with syndromic IDDs present with medically complex phenotypes and remain minimally verbal even into adulthood, creating challenges accessing and benefiting from community-based interventions. Several reports point to extreme behavior and communication challenges as the most pressing behavioral health concerns for this population. Our near-term goals seek to identify effective approaches to target the more severe cognitive and behavioral phenotypes found in syndromic IDDs. Here, we propose adaptations to function-based treatment (FBT) – an already well-established, person- centered applied behavior analysis (ABA) model focused on replacing challenging behaviors with prosocial communication and behavior responses. Using the Planned Adaptation approach, proactive adaptations to improve the fit of FBT with the syndromic IDD population include syndrome-specific characterizations to inform phenotype-environment interactions, systematic screening for automatically reinforced behaviors which are often excluded from published FBT approaches, and adjustments to support minimally verbal individuals. This proposal draws upon the expertise of the investigative group in syndromic IDDs, conventional and telehealth behavioral interventions, and implementation sciences to evaluate the adapted, parent-implemented, telehealth FBT model for syndromic IDDs (FBTsIDD). The goal of this fully remote hybrid type 1 effectiveness- implementation study is to test FBTsIDD as delivered by non-specialist providers housed in medical hubs serving individuals with syndromic IDDs. Aim 1 involves a 24-week randomized control trial (RCT) that will randomize 80 children (ages 2 to 12 years) with syndromic IDDs and moderate to severe intellectual disability (ID) and their caregivers and randomize them into FBTsIDD or positive-parenting treatment (Treatment as Usual, TAU). Our overarching hypothesis is that FBTsIDD will be associated with significant reductions in challenging behaviors compared to TAU on independent evaluator ratings using a consumer-driven, Parent Target Problems (PTP) inventory and standardized measures of behavior and functional communication. Aim 2 seeks to systematically measure and understand both planned and unplanned adaptions to FBTsIDD using the Framework for Reporting Adaptations and Modifications-Expanded (FRAME). Together, these aims provide an innovative model to develop effective, acceptable, and scalable interventions for behavioral and communication challenges across the diverse, vulnerable population of individuals with syndromic IDDs.

项目概要/摘要： 2019冠状病毒病服务中断对智力和发展方面的个人的巨大影响 国际残疾人日强调，迫切需要增加获得精神和行为健康的机会 服务在缺碘症人群中，与缺碘症相关的遗传综合征（“综合征性缺碘症”）代表了一种 尤其是弱势群体。许多IDDs综合征患者出现医学上复杂的 表型和保持最低限度的语言，甚至到成年，创造挑战，访问和受益 从基于社区的干预措施。几份报告指出了极端行为和沟通方面的挑战 是这一人群最迫切的行为健康问题。我们的近期目标是确定 针对综合征型IDDs中发现的更严重的认知和行为表型的有效方法。 在这里，我们建议适应功能为基础的治疗（FBT）-一个已经建立良好的，人- 中心应用行为分析（阿坝）模型侧重于用亲社会行为取代挑战行为 沟通和行为反应。采用有计划的适应方法，积极主动地适应 提高FBT与综合征型IDD人群的拟合度，包括综合征特异性特征， 表型-环境相互作用，系统筛选自动强化的行为， 通常被排除在已公布的FBT方法之外，并进行调整以支持最低限度的口头个体。这 该提案借鉴了综合征型碘缺乏病、常规和远程保健调查小组的专业知识 行为干预和实施科学，以评估适应，家长实施，远程保健 综合征型IDD的FBT模型（FBTsIDD）。这种完全远程混合1型有效性的目标- 实施研究的目的是测试由医疗中心的非专家提供者提供的FBTsIDD， IDDs综合征患者。目标1涉及一项为期24周的随机对照试验（RCT）， 80名患有综合征型IDDs和中度至重度智力残疾（ID）的儿童（2至12岁）， 他们的照顾者，并将他们随机分为FBTsIDD或积极的父母治疗（治疗作为母亲，TAU）。 我们的总体假设是，FBTsIDD将与挑战性疾病的显著减少相关。 使用消费者驱动的父目标， 问题（PTP）库存和行为和功能沟通的标准化措施。目标2寻求 使用系统性测量和了解对FBTsIDD的计划内和计划外适应 报告调整和修改框架（FRAME）。总之，这些目标提供了一个 创新模式，以制定有效的，可接受的和可扩展的干预措施， 在患有综合征型缺碘症的各种脆弱人群中，沟通面临挑战。