Behavioral Inflexibility in IDD Outcome Measurement

IDD 结果测量中的行为僵化

• 批准号: 8801467
• 负责人: Brian Antonio Boyd
• 金额: $ 65.15万
• 依托单位: UNIV OF NORTH CAROLINA CHAPEL HILL
• 依托单位国家: 美国
• 财政年份: 2014
• 资助国家: 美国
• 起止时间: 2014-12-10 至 2019-11-30
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/8801467
• 关键词: Activities of Daily Living; Adolescent; Affective; Age; Autistic Disorder; Behavior; Behavioral; Behavioral trial; Characteristics; Child; Clinical; Clinical Trials; Cognitive; Conduct Clinical Trials; Consensus; Data; Development; Device or Instrument Development; Distress; Emotional; Environment; Equipment and supply inventories; Family; Focus Groups; Fragile X Syndrome; Funding; Genetic; Goals; Hereditary Disease; Impairment; Intellectual functioning disability; Intervention; Lead; Learning; Life; Link; Measurement; Measures; Methodology; Moods; Movement; Outcome; Outcome Measure; Parents; Persons; Pharmaceutical Preparations; Physical therapy exercises; Population; Positioning Attribute; Prader-Willi Syndrome; Problem behavior; Property; Psychometrics; Quality of life; Reporting; Request for Applications; Research; Research Design; Research Personnel; Rett Syndrome; Sampling; Self-Injurious Behavior; Stereotyping; Structure; Surveys; Symptoms; Syndrome; Temperament; Testing; Time; Translational Research; Treatment Efficacy; Treatment outcome; autism spectrum disorder; behavioral impairment; compulsion; design; experience; falls; flexibility; instrument; interest; novel; pill; public health relevance; sound; stem; therapy development

DESCRIPTION (provided by applicant): This RO1 application requests funds to develop and validate a novel outcome measure targeting the behavioral inflexibility in children with intellectual and developmental disabilities (IDD). This behavioral domain encompasses a host of closely related clinical sequelae that can be profoundly disabling and are common in IDD such as (a) an overall inflexible temperament or behavioral style that underlies discrete idiosyncratic forms of repetitive and rigid behavior manifestations (e.g., inability to tolerate changes in the environment, stereotyped uses of objects); (b) associated mood and behavior problems that arise when this inflexible temperament and its manifestations cannot be accommodated; and (c) interference with learning and adaptation that stems from rigidity / inflexibility as even treatmen itself can represent intrusion and change for an inflexible child (e.g., a new pill, a new therapy exercise). In recent years there have been large-scale drug trials and small behavioral trials targeting discrete behaviors that fall under the domain of repetitive inflexible behavior. However, the measures included in many of these clinical trials have little to no evidence of their ability o detect change over time. In fact, while several instruments exist that measure the discrete behavioral endpoints of this overall construct of rigidity / inflexibility, no instrument exists tht adequately extends much beyond a "behavior checklist" approach. The overall objective of this application is to develop and validate a new measure, the Repetitive and Inflexible Behavior Inventory (RIBI), with the primary goal of developing a measure that is sensitive to change over time. The RIBI is designed as a parent report measure for children (ages 3 -18) with IDD. The rationale is that a novel measure is needed to (a) capture the range of repetitive inflexible behaviors that are found in children with IDD across a variety of idiopathic and genetic disorders, (b) measure the functional impact of these behaviors on the family's daily life, and (c) detect change over time when these behaviors are the target of intervention. Thus, the specific aims of this project are to (1) examine the psychometric properties of the RIBI for children and adolescents with autism spectrum disorder (ASD), establishing reliability, factor structure, content and construct validity; (2) evaluate the RIBI's ability to measure change over time in children with ASD; and (3) examine the psychometric stability of the RIBI when applied to children and adolescents with Fragile-X and Prader-Willi syndromes. To accomplish these aims, a multisite study will be conducted using recommended instrument development methodologies, such as focus groups, expert panels, and online surveys (n=1500). This team is in an ideal position to develop the RIBI because of our prior experience with measure development, and the conduct of clinical trials targeting repetitive inflexible behavior. The expected deliverable i an outcome measure that is appropriate for IDD populations and minimally burdensome to complete in order to be useful for multisite clinical trials.

描述(由申请人提供)：此RO1申请申请资金，以开发和验证一项针对智力和发育障碍(IDD)儿童行为不灵活的新结果衡量标准。这一行为领域包含一系列密切相关的临床后遗症，这些后遗症在IDD中很常见，例如(A)总体僵化的气质或行为风格，其背后是离散的重复和僵化的行为表现形式(例如，无法容忍环境的变化，刻板地使用物体)；(B)当这种僵化的气质及其表现无法适应时产生的相关情绪和行为问题；以及(C)由于僵硬/僵硬导致的学习和适应干扰，即使对待男性本身也可能代表着对僵硬的儿童的侵扰和改变(如新的药片、新的治疗练习)。近年来，已经有大规模的药物试验和针对离散行为的小型行为试验，这些行为属于重复僵化行为的领域。然而， 许多临床试验中包含的测量方法很少或根本没有证据表明它们能够随着时间的推移而检测变化。事实上，虽然有几种工具可以衡量僵化/僵化这一整体结构的离散行为端点，但没有一种工具可以充分扩展到“行为核对表”方法之外。此应用程序的总体目标是开发和验证一种新的测量方法，即重复和僵化行为清单(RIBI)，其主要目标是开发一种对随时间变化敏感的测量方法。RIBI是为患有IDD的儿童(3-18岁)设计的家长报告测量工具。理由是需要一种新的措施来(A)捕捉IDD儿童在各种特发性和遗传性疾病中发现的重复的僵化行为的范围，(B)衡量这些行为对家庭日常生活的功能影响，以及(C) 当这些行为成为干预目标时，检测随时间的变化。因此，本项目的具体目标是(1)检验RIBI对患有自闭症谱系障碍(ASD)的儿童和青少年的心理测量学特性，建立信度、因素结构、内容和结构效度；(2)评估RIBI测量ASD儿童随时间变化的能力；以及(3)检验RIBI应用于患有Fragile-X和Prader-Willi综合征的儿童和青少年时的心理测量学稳定性。为实现这些目标，将使用建议的工具开发方法，如焦点小组、专家小组和在线调查(n=1500)，进行多点研究。这个团队处于开发RIBI的理想位置，因为我们以前在测量开发方面的经验，以及针对重复的僵硬行为进行的临床试验。预期可交付成果是一种适合于IDD人群的结果衡量标准，完成时负担最小，以便对多点临床试验有用。