Expressive Language Sampling as an Outcome Measure

表达性语言抽样作为结果衡量标准

• 批准号: 8811461
• 负责人: LEONARD J. ABBEDUTO
• 金额: $ 59.13万
• 依托单位: UNIVERSITY OF CALIFORNIA AT DAVIS
• 依托单位国家: 美国
• 财政年份: 2013
• 资助国家: 美国
• 起止时间: 2013-03-01 至 2016-02-29
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/8811461
• 关键词: Address; Adolescent and Young Adult; Age; Animal Model; Area; Autistic Disorder; Behavior; Behavior Therapy; Behavioral; Berry; Child; Clinical; Clinical Trials; Code; Cognitive; Communities; Development; Diagnostic; Dimensions; Disease; Down Syndrome; Etiology; Evaluation; Fragile X Syndrome; Gender; Genetic; Genetic Transcription; Health; Heterogeneity; Human; Impairment; Individual; Institutes; Intellectual functioning disability; Language; Longevity; Measures; Molecular Genetics; Narration; Nature; Neurocognitive; Outcome Measure; Parents; Participant; Pharmacologic Substance; Pharmacological Treatment; Phase; Phenotype; Population; Procedures; Process; Property; Psychometrics; Recording of previous events; Recruitment Activity; Reporting; Research; Role; Sampling; Schedule; School-Age Population; Severities; Site; Structure; Symptoms; System; Techniques; Testing; Time; Translations; Treatment Efficacy; Ursidae Family; Variant; clinical practice; clinically relevant; experience; follow-up; indexing; informant; neuromechanism; psychosocial; skills; syntax; treatment effect

DESCRIPTION (provided by applicant): New treatments for people with intellectual disabilities (ID) are increasingly condition-specific in nature. Numerous clinical trials of targeted pharmacological agents are now in process for fragile X syndrome (FXS) and Down syndrome (DS). Condition-specific behavioral treatments also are emerging. Evaluation of all such treatments is being hampered by a lack of adequate cognitive and behavioral endpoints. In this project, we propose to evaluate the adequacy of expressive language sampling for deriving language-relevant clinical endpoints. In this procedure, expressive language samples are collected in highly structured and scripted, yet naturalistic, interactions. These samples can then be analyzed to derive clinical endpoints reflecting important dimensions of language skill and atypical language behavior. Although the suitability of expressive language sampling for clinical trials with FXS or DS (or ID more generally) has yet to be determined, the procedures are especially promising because they yield clinically relevant and functional endpoints, have been shown to capture impairments that are common to ID as well as those specific to FXS or DS, and have been shown to yield robust indicators of developmental change within typical and other language-impaired populations. In this project, we propose: (1) to examine the basic psychometric properties of measures derived from expressive language sampling techniques, including establishing their test-retest reliability, internal consistency, validity, and sensitiviy; (2) to evaluate differences in the psychometric properties of expressive language sampling techniques as a function of variations in participant etiology, age, gender, autism symptom severity, and level of ID; (3) to compare the psychometric properties of three different expressive language sampling techniques; and (4) to evaluate the feasibility of implementing the expressive language sampling across multiple sites, as would be required in a typical clinical trial. These aims will be addressed by collecting expressive language samples from children, adolescents, and young adults with FXS or DS. Samples will be collected within three interaction formats: conversation, narration, and the structured interactions comprising the Autism Diagnostic Observation Schedule (ADOS). Measures derived from the samples will include those indexing syntax (an area of especially severe impairment in DS) and perseveration (an area of especially severe impairment for FXS). Test-retest reliability will be assessed at 4 weeks (+/- 1 week) using alternate versions of sampling materials. Internal consistency will be assessed by computing alpha coefficients within and across sampling techniques. Standardized tests and informant report will be used as indicators of validity. A two-year longitudinal follow-up will yield an estimate of sensitivity to change. Participants will be tested at multiple sites, each with considerable experience in the evaluation of individuals with FXS or DS. Feasibility of multiple-site implementation will be evaluated by comparing language samples across sites on key indicators. Transcription, coding, and analysis will be conducted only at the UC Davis MIND Institute site.

描述（由申请人提供）：针对智障人士（ID）的新疗法本质上越来越特定。现在，针对靶向药理剂的大量临床试验正在进行脆弱的X综合征（FXS）和唐氏综合症（DS）的过程。条件特定的行为治疗也正在出现。缺乏足够的认知和行为终点，对所有此类治疗的评估都受到了阻碍。在这个项目中，我们建议评估表达语言抽样的适当性，以推导与语言相关的临床终点。在此过程中，表达语言样本是在高度结构化和脚本却具有自然主义的相互作用中收集的。这些样本可以 进行分析以得出临床终点，以反映语言技能和非典型语言行为的重要方面。尽管尚未确定表达语言对使用FXS或DS（或ID）进行临床试验的适用性，但这些程序尤其有希望，因为它们在临床上相关且功能性的终点，已证明可以捕获与ID相同的损害，并且与FXS或DS相比，并且已证明了这些损害以及在FXS或DS的范围内均可构成众多范围内的范围和其他语言范围。在该项目中，我们建议：（1）检查来自表达语言采样技术的度量的基本心理测量特性，包括建立其测试可靠性，内部一致性，有效性和敏感性； （2）评估表达语言抽样技术的心理测量特性差异，这是参与者病因，年龄，性别，自闭症症状严重程度和ID水平的差异的函数； （3）比较三种不同表达语言抽样技术的心理测量特性； （4）评估在典型的临床试验中所要求的，以评估多个站点跨多个站点实施表达语言采样的可行性。这些目标将通过收集具有FXS或DS的儿童，青少年和年轻人的表达性语言样本来解决。样本将在三种交互格式中收集：对话，叙述以及包括自闭症诊断观察时间表（ADO）的结构化相互作用。从样品中得出的措施将包括那些索引语法（DS中特别严重的损害区域）和持久性（FXS特别严重损害的区域）。重测可靠性将在4周（+/- 1周）使用替代版本的采样材料评估。内部一致性将通过计算在抽样技术内部和跨采样技术内的α系数来评估。标准化测试和线人报告将用作有效性的指标。两年的纵向随访将产生对变化敏感性的估计。参与者将在多个站点进行测试，每个站点在评估FXS或DS的人方面具有丰富的经验。通过比较关键指标上的跨站点的语言样本来评估多个站点实现的可行性。转录，编码和分析仅在UC Davis Mind Institute站点进行。