Telehealth-delivered outcome measures for Spanish- and English-speaking people with Down syndrome

远程医疗为讲西班牙语和英语的唐氏综合症患者提供结果测量

• 批准号: 10730307
• 负责人: LEONARD J. ABBEDUTO
• 金额: $ 44.13万
• 依托单位: UNIVERSITY OF CALIFORNIA AT DAVIS
• 依托单位国家: 美国
• 财政年份: 2023
• 资助国家: 美国
• 起止时间: 2023-09-20 至 2025-08-31
• 项目状态: 未结题
• 来源: https://reporter.nih.gov/project-details/10730307
• 关键词: Acceleration; Address; Adolescent; Adolescent and Young Adult; Adult; Age; Aging; Alzheimer' s Disease; Alzheimer' s disease risk; Articulation; Behavioral; Berry; Birth; Books; Brain; Caregivers; Censuses; Child; Childhood; Clinic; Clinical Trials; Cognitive; Data; Development; Down Syndrome; Elderly; Ensure; Equity; Evaluation; Family; Future; General Population; Genetic; Growth; Health Services Accessibility; Home; Impairment; Individual; Intellectual functioning disability; Intervention; Language; Late Onset Alzheimer Disease; Life; Life Expectancy; Linguistics; Link; Longevity; Measures; Mental Health; Methods; Narration; Outcome Measure; Participant; Performance; Persons; Pharmaceutical Preparations; Phenotype; Population; Prevalence; Procedures; Property; Psychometrics; Research; Sample Size; Sampling; Speech; Standardization; Symptoms; Testing; Training; Travel; Treatment Efficacy; Validation; Vocabulary; Work; Youth; autism spectrum disorder; bilingualism; cognitive function; daily functioning; efficacy evaluation; efficacy testing; emerging adult; executive function; expectation; health training; high risk; improved; indexing; individuals with autism spectrum disorder; language outcome; neuropathology; non-verbal; patient home care; pilot test; prevent; recruit; sound; spatial memory; syntax; telehealth; therapy design; verbal; virtual

ABSTRACT Down syndrome (DS) is the leading genetic cause of intellectual disability. Moreover, virtually all aspects of cognitive and behavioral development show very slow rates of development relative to age expectations in people with DS. Life expectancy for individuals with DS has increased in recent decades; however, DS is also characterized by early aging-related declines and a high risk for Alzheimer’s Disease (AD). Thus, there is a need in the DS field to develop treatments that accelerate development during childhood and beyond as well as treatments that slow or prevent the inevitable declines associated with AD. Evaluating the efficacy of such treatments, however, requires psychometrically adequate measures of change. The Expressive Language Sampling-Narrative (ELS-N) procedure has been shown to be a psychometrically sound measure for tracking expressive language growth with age or within treatment in children, adolescents, and young adults with DS. There is also preliminary evidence that ELS-N is sensitive to some early signs of decline. However, ELS-N has not been validated in older adults with DS; has not been evaluated for non-English speakers; and has been tested only in a clinic-based, clinician-administered format. To make ELS-N accessible to a wider array of diverse participants, we have developed, and pilot tested, English and Spanish versions of ELS-N that can be administered by caregivers to individuals with autism at home after telehealth-delivered training to caregivers. In the proposed project, we further expand ELS-N by recruiting 60 verbal individuals with DS, ages 10 to 40 years, along with their caregivers, with equal numbers of English and Spanish speakers. Two ELS-N delivery methods are proposed: (a) at-home, caregiver-implemented (after telehealth training) and (b) the “classic” version of ELS-N (i.e., in-clinic, professional-administered). Each participant with DS will receive the two ELS-N delivery methods. The specific aims of the project are: (1) examine the consistency of performance individuals with DS across the two ELS-N delivery methods; (2) examine practice effects, test-retest reliability, and construct validity for the variables derived from each ELS-N delivery method; and (3) examine the consistency of performance and psychometric properties of the ELS-derived variables separately for ages 10-20, 21-30, and 31-40 years, although this last aim is exploratory due to limited sample size. The aims will be addressed for the sample as a whole and for each language group separately, thereby providing data relevant to future studies involving either multi-language or single-language samples of participants. This project will expand the array of outcome measures available for assessing treatment efficacy across the lifespan of individuals with DS, increase the potential for broader representation in research and improve equitable access to care, and identify potential linguistic indicators of both growth and decline.

抽象的 唐氏综合症（DS）是智力障碍的主要遗传原因。而且，几乎所有方面 认知和行为发展相对于年龄期望的发展速度非常缓慢 患有DS的人。近几十年来，DS个人的预期寿命有所增加。但是，DS也是 其特征是早期衰老相关的下降和阿尔茨海默氏病（AD）的高风险。那有一个 在DS领域的需求，以开发在童年及以后加速发展的治疗方法 作为缓慢或阻止与AD相关的不可避免下降的治疗方法。评估这种效率 但是，治疗需要在心理上足够的变化措施。富有表现力的语言 样本叙事（ELS-N）程序已被证明是跟踪的一种心理良好的措施 随着年龄或在儿童，青少年和DS的年轻人的治疗中，随着年龄的年龄或治疗中的表现性语言增长。 也有初步证据表明Els-N对一些早期衰落的迹象敏感。但是，Els-N有 在患有DS的老年人中未得到验证；尚未对非英语说话者进行评估；一直是 仅在基于临床的临床管理格式中进行测试。使Els-n可以访问一系列 潜水员参与者，我们已经开发并进行了试验测试，ELS-N的英语和西班牙语版本可以是 在对护理人员进行远程医疗培训后，由护理人员管理自闭症患者。 在拟议的项目中，我们通过招募10至40岁的DS口头个体进一步扩展ELS-N 多年来，与他们的看护人，英语和西班牙语的人相等。两个ELS-N交付 提出了方法：（a）在家，照顾者实施（远程医疗培训之后）和（b）“经典” ELS-N的版本（即，临床，专业的管理）。每个具有DS的参与者将获得两个ELS-N 递送方法。该项目的具体目的是：（1）检查性能个人的一致性 在两种ELS-N输送方法中具有DS； （2）考试实践效果，测试可靠性和 从每种ELS-N输送方法得出的变量的构造有效性； （3）检查一致性 10-20、21-30岁的ELS衍生变量的性能和心理测量特性 和31 - 40年，尽管由于样本量有限，探索了最后一个目标。目标将被解决 对于整个样本和每个语言组的样本，从而提供与未来相关的数据 涉及参与者多语言或单语言样本的研究。该项目将扩大 可用于评估个人寿命的治疗效率的一系列结果指标 DS，增加在研究中更广泛代表的潜力，并提高公平的护理访问权限，并 确定增长和下降的潜在语言指标。