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

摘要 唐氏综合症(DS)是导致智力残疾的主要遗传原因。此外，几乎所有方面的 认知和行为发育显示出相对于年龄预期非常缓慢的发育速度 多发性硬化症患者。近几十年来，患有DS的人的预期寿命有所增加；然而，DS也 以早期衰老相关的衰退和阿尔茨海默病(AD)的高风险为特征。因此，有一个 需要在DS领域开发促进儿童时期及以后发育的治疗方法 作为减缓或防止与阿尔茨海默病相关的不可避免的下降的治疗方法。评估这种方法的效果 然而，治疗需要心理测量学上足够的改变措施。富有表现力的语言 抽样-叙述(ELS-N)程序已被证明是一种心理测量学上良好的跟踪措施 在患有DS的儿童、青少年和年轻成人中，随着年龄的增长或在治疗期间，语言表达能力的增长。 也有初步证据表明，ELS-N对一些早期的下降迹象很敏感。然而，ELS-N拥有 没有在患有DS的老年人中得到验证；没有在非英语使用者中进行评估；并且已经 仅在以临床为基础的、临床医生管理的形式下进行测试。使更多的用户能够访问ELS-N 不同的参与者，我们开发了ELS-N的英语和西班牙语版本，并进行了试点测试，可以 由照顾者在远程健康向照顾者提供培训后，在家中对自闭症患者进行管理。 在拟议的项目中，我们通过招募60名患有DS的口头患者来进一步扩展ELS-N，年龄在10到40岁之间 几年，以及他们的照顾者，讲英语和西班牙语的人数相同。两个ELS-N交付 建议的方法是：(A)在家中由照顾者实施(在远程保健培训之后)和(B)“经典” ELS-N版(即，门诊、专业管理)。每个有DS的参与者将收到两个ELS-N 投递方式。该项目的具体目标是：(1)检查绩效个体的一致性 在两种ELS-N交付方法中使用DS；(2)检查练习效果、重测可靠性和 对每种ELS-N交付方法得出的变量的结构效度；以及(3)检验一致性 ELS衍生变量在10-20岁、21-30岁、 31年至40年，尽管由于样本量有限，最后一个目标是探索性的。这些目标将得到解决 作为一个整体的样本和单独的每种语言组，从而提供与未来相关的数据 研究涉及参与者的多语言样本或单一语言样本。该项目将扩大 一系列可用于评估慢性阻塞性肺疾病患者终身治疗效果的结果衡量标准 DS，增加研究中更广泛代表性的可能性，改善公平获得护理的机会，以及 确定潜在的语言指标，既有增长，也有下降。

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.