Exploring the validity of articulatory impairment phenotypes in speech motor disorders

探索言语运动障碍中发音障碍表型的有效性

• 批准号: 10331841
• 负责人: Hannah Prescott Rowe
• 金额: $ 1.95万
• 依托单位: MGH INSTITUTE OF HEALTH PROFESSIONS
• 依托单位国家: 美国
• 财政年份: 2021
• 资助国家: 美国
• 起止时间: 2021-01-20 至 2022-08-02
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/10331841
• 关键词: Acoustics; Address; Amyotrophic Lateral Sclerosis; Apraxias; Area; Articulation; Behavioral; Biological Markers; Clinical; Clinical Trials; Communication; Communication impairment; Development; Diagnosis; Discriminant Analysis; Disease; Dysarthria; Ensure; Functional disorder; Goals; Impairment; Individual; Intervention; Knowledge; Language; Link; Measures; Mission; Monitor; Motion; Motor; Motor Neurons; National Institute on Deafness and Other Communication Disorders; Nerve Degeneration; Neurologic; Outcome Measure; Parkinson Disease; Patient-Focused Outcomes; Patients; Performance; Pharmacologic Substance; Phenotype; Population; Primary Progressive Aphasia; Progressive Nonfluent Aphasias; Regression Analysis; Repetitive Sequence; Research; Research Personnel; Research Priority; Series; Severities; Speech; Speed; Spinocerebellar Ataxias; Strategic Planning; Testing; Treatment Efficacy; Treatment outcome; Voice; Work; base; behavioral phenotyping; clinical phenotype; clinical practice; efficacy evaluation; falls; improved; indexing; individualized medicine; innovation; motor control; motor deficit; motor disorder; motor impairment; novel; patient response; personalized medicine; rehabilitation research; speech accuracy; tool; treatment response; treatment strategy

PROJECT SUMMARY Speech motor disorders have profound impacts on an individual’s ability to communicate, often leading to a significant reduction in quality of life35,49. Since the advent of personalized medicine, clinical phenotypes have become an increasingly important construct in rehabilitation research, as they facilitate the identification of treatment targets that are individualized to a patient’s unique impairment profile3. There is, however, currently no established set of objective measures that to phenotype the articulation impairments observed in speech motor disorders32. Consequently, clinicians employ broad treatment strategies for patients with distinct articulatory deficits, which often result in variable therapy outcomes97. Given the links between specific articulatory abnormalities and pathophysiologies1,16,81,90 and the impact of the articulatory subsystem on intelligibility14,53,69,77, there is a critical need to determine the articulatory impairment phenotypes across the spectrum of speech motor disorders. The proposed study will comprehensively characterize articulatory impairments in four neurologic populations (compared to healthy controls) with hypothetically divergent motor deficits: (1) the nonfluent variant of primary progressive aphasia (nfvPPA) or primary progressive apraxia of speech (PPAOS), (2) amyotrophic lateral sclerosis (ALS), (3) Parkinson’s disease (PD), and (4) spinocerebellar ataxia (SCA). Articulatory impairments will be characterized based on a hypothesis-driven framework of motor control (i.e., Coordination, Consistency, Speed, Precision, and Rate) composed of a semi-automated acoustic feature set. Aim 1 will use a linear discriminant analysis (LDA) to compare the articulatory performance (as indexed by the acoustic feature set) of the groups during the sequential motion rate (SMR) task. The LDA will be adjusted for speech severity to determine the true discriminatory power of the acoustic features and ensure that severity differences are not driving phenotype differences. Aim 2 will use a multiple regression analysis (MRA) to determine the articulatory deficits most associated with intelligibility in the four neurologic populations by correlating performance on each acoustic feature with performance on the Sentence Intelligibility Test (SIT)96. The overall goal of the proposed research is to advance our knowledge of the diversity of articulatory impairment phenotypes in different speech motor subtypes. Results from this research will (1) facilitate the development of impairment-based approaches, (2) yield more granular outcome measures for evaluating the efficacy of behavioral or pharmaceutical treatments, and (3) elucidate the contribution of distinct articulatory mechanisms to declines in functional communication. This project falls under NIDCD’s Priority Area 3 in Voice, Speech, and Language Research, as it investigates biomarkers that could support diagnosis, treatment, and progress monitoring in individuals with speech impairments. Furthermore, this work is closely aligned with the strategic plan for behavioral phenotyping and is overall consistent with the mission of NIDCD to further our knowledge and understanding of communication disorders.

项目概要 言语运动障碍对个人的沟通能力有着深远的影响，通常会导致 自从个性化医疗出现以来，生活质量显着下降35,49。 成为康复研究中越来越重要的结构，因为它们有助于识别 然而，目前存在针对患者独特损伤情况的个性化治疗目标3。 没有一套既定的客观措施来对言语中观察到的发音障碍进行表型分析 运动障碍 32. 鉴于特定之间的联系，发音缺陷通常会导致不同的治疗结果97。 关节异常和病理生理学1,16,81,90以及关节子系统对 智力14,53,69,77，迫切需要确定整个群体的发音障碍表型 拟议的研究将全面描述言语运动障碍的特征。 四个神经系统群体（与健康对照相比）存在假设运动不同的损伤 缺陷：（1）原发性进行性失语症（nfvPPA）的不流利变体或原发性进行性失用症 言语 (PPAOS)、(2) 肌萎缩侧索硬化症 (ALS)、(3) 帕金森病 (PD) 和 (4) 脊髓小脑性共济失调 (SCA) 将根据假设驱动进行表征。 电机控制框架（即协调性、一致性、速度、精度和速率）由 目标 1 将使用线性判别分析 (LDA) 来比较半自动声学特征集。 顺序运动过程中各组的发音表现（按声学特征集索引） LDA 将根据语音严重程度进行调整，以确定真实的歧视能力。 声学特征并确保严重性差异不会导致表型差异。目标 2 将使用 多元回归分析（MRA）以确定与可懂度最相关的发音缺陷 通过将每个声学特征的表现与 句子清晰度测试（SIT）96。拟议研究的总体目标是增进我们的知识。 不同言语运动亚型中发音障碍表型的多样性。 研究将（1）促进基于损伤的方法的开发，（2）产生更精细的结果 评估行为或药物治疗功效的措施，以及（3）阐明 不同的发音机制导致功能性沟通下降的原因属于该项目。 NIDCD 的优先领域 3 是语音、言语和语言研究，因为它研究的生物标志物可以 支持有言语障碍的个体的诊断、治疗和进展监测。 这项工作与行为表型分析的战略计划密切相关，总体上与 NIDCD 的使命是增进我们对沟通障碍的认识和理解。