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RO 1申请要求资金开发和验证一种针对智力和发育障碍（IDD）儿童行为能力的新型结局指标。这一行为领域包括许多密切相关的临床后遗症，这些后遗症可能严重致残，并且在IDD中很常见，例如（a）作为重复和僵化行为表现的离散特异质形式的基础的整体僵化气质或行为风格（例如，不能容忍环境的变化，对物体的刻板使用）;（B）当这种不灵活的气质及其表现不能被适应时出现的相关的情绪和行为问题;以及（c）源于僵化/不灵活的学习和适应的干扰，因为即使对待本身也可以代表对不灵活的孩子的侵入和改变（例如，一种新的药丸，一种新的治疗练习）。近年来，已经有大规模的药物试验和小规模的行为试验，目标是属于重复性刚性行为领域的离散行为。然而，在这方面， 许多这些临床试验中包括的测量几乎没有证据表明它们能够检测随时间的变化。事实上，虽然存在几种工具来测量这种刚性/可扩展性的整体结构的离散行为端点，但没有工具能够充分扩展到“行为检查表”方法之外。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是为患有碘缺乏病的儿童（3 - 18岁）设计的父母报告措施。其基本原理是需要一种新的测量方法来（a）捕获IDD儿童在各种特发性和遗传性疾病中发现的重复性不灵活行为的范围，（B）测量这些行为对家庭日常生活的功能影响，以及（c） 当这些行为是干预的目标时，检测随时间的变化。因此，本研究的具体目的是：（1）检验RIBI对自闭症谱系障碍（ASD）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）检查RIBI应用于患有脆性X和Prader-Willi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中心研究，例如焦点小组、专家小组和在线调查（n=1500）。这个团队是一个理想的位置，因为我们以前的经验，衡量发展，并针对重复的不灵活的行为进行临床试验，以开发RIBI。预期可交付成果是一种结局指标，适用于IDD人群，完成负担最小，以便对多中心临床试验有用。