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 申请要求资金来开发和验证针对智力和发育障碍 (IDD) 儿童行为僵化的新颖结果测量。这一行为领域包含一系列密切相关的临床后遗症，这些后遗症可能会造成严重的残疾，并且在 IDD 中很常见，例如： (b) 当无法适应这种顽固的气质及其表现时，就会出现相关的情绪和行为问题； (c) 由于僵化/不灵活而对学习和适应的干扰，因为即使是治疗本身也可能代表对不灵活的儿童的侵入和改变（例如，新药、新的治疗练习）。近年来，出现了针对重复性不灵活行为领域的离散行为的大规模药物试验和小型行为试验。然而， 许多临床试验中包含的措施几乎没有证据表明它们有能力检测随时间的变化。事实上，虽然存在多种工具来测量这种僵化/不灵活的整体结构的离散行为端点，但没有一种工具能够充分扩展“行为检查表”方法之外的范围。该应用程序的总体目标是开发和验证一种新的测量方法，即重复和不灵活行为量表 (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 旨在作为 IDD 儿童（3 -18 岁）的家长报告衡量标准。理由是，需要一种新的措施来（a）捕获在各种特发性和遗传性疾病的 IDD 儿童中发现的重复性僵化行为的范围，（b）测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，该项目的具体目标是（1）检查 RIBI 对自闭症谱系障碍（ASD）儿童和青少年的心理测量特性，建立可靠性、因素结构、内容和结构有效性； (2) 评估 RIBI 衡量 ASD 儿童随时间变化的能力； (3) 检查 RIBI 在应用于患有脆性 X 综合征和普瑞德威利综合征的儿童和青少年时的心理测量稳定性。为了实现这些目标，将使用推荐的工具开发方法进行多地点研究，例如焦点小组、专家小组和在线调查 (n=1500)。由于我们之前在测量开发以及针对重复性不灵活行为的临床试验方面的经验，该团队处于开发 RIBI 的理想位置。预期交付成果是一种结果衡量标准，适合 IDD 人群，并且完成起来的负担最小，以便可用于多中心临床试验。