Behavioral Inflexibility in IDD Outcome Measurement

IDD 结果测量中的行为僵化

• 批准号: 9180065
• 负责人: Brian Antonio Boyd
• 金额: $ 60.02万
• 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/9180065
• 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; Exercise Therapy; 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; 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; compulsion; design; experience; falls; flexibility; instrument; interest; novel; novel therapeutics; pill; public health relevance; repetitive behavior; sound; stem

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申请要求资金制定和验证针对智力和发育障碍儿童（IDD）行为不灵活的新成果措施（IDD）。这个行为领域涵盖了许多密切相关的临床后遗症，这些后遗症可能非常残疾，并且在IDD中很常见，例如（a）（a）总体不灵活的气质或行为风格，这些气质或行为风格是基于分散的重复性和僵化行为表现形式的特殊形式的基础（例如，无法在环境中耐受性，对象使用了对象的使用，可以容忍在环境中的变化）； （b）当无法适应这种僵化的气质及其表现时会出现相关的情绪和行为问题； （c）干扰源于刚性 /不灵活的学习和适应，因为即使是治疗疗法本身也可以代表不灵活的儿童的入侵和变化（例如，一种新药丸，一种新的疗法运动）。近年来，已经进行了大规模的药物试验和针对离散行为的小型行为试验，这些试验属于重复性不灵活行为的领域。然而， 许多这些临床试验中包括的措施几乎没有证据表明其能力o随着时间的推移检测变化。实际上，尽管存在几种仪器，这些仪器衡量了这种刚性 /僵化性整体结构的离散行为终点，但没有任何仪器可以充分地扩展到“行为清单”方法之外。该应用程序的总体目的是开发和验证一种新的措施，重复性和僵化的行为清单（RIBI），其主要目标是开发一种对随着时间的变化而敏感的度量。 RIBI被设计为带有IDD的儿童（3 -18岁）的父级报告措施。理由是（a）需要一种新的措施来捕获在各种特发性和遗传性疾病中IDD儿童中发现的重复不灵活行​​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） 当这些行为是干预的目标时，随着时间的推移检测变化。因此，该项目的具体目的是（1）检查自闭症谱系障碍儿童和青少年RIBI的心理测量特性（ASD），确定可靠性，因子结构，内容和构造有效性； （2）评估RIBI在ASD儿童中测量随时间变化的能力； （3）当将RIBI的心理测量稳定性应用于患有脆弱的X和Prader-Willi综合征的儿童和青少年时。为了实现这些目标，将使用推荐的仪器开发方法（例如焦点小组，专家小组和在线调查）进行多站点研究（n = 1500）。由于我们先前在测量开发方面的经验以及针对重复不灵活行​​为的临床试验的进行，该团队处于发展RIBI的理想位置。预期的可交付i是适合IDD人群和最小繁重的结果的结果措施，以便对多站点临床试验有用。