Validation of an automated acoustic outcome measure for AdLD

项目摘要 喉肌张力障碍（LD）是一种神经性语音障碍，其特征是喉的不自主痉挛 肌肉在发声，这显着影响心理健康和生活质量。最 LD的常见亚型是喉内收肌张力障碍（AdLD），其涉及喉肌的痉挛 使声带内收导致LD不连续。LD不连续是对语音的干扰 由痉挛引起的声音信号，包括发声中断（语音中断）和频率偏移（音高 在发声过程中最严重的中断）。治疗AdLD的一个重要问题是， 误诊AdLD经常被误认为是肌肉紧张性发声障碍（MTD），这是一种功能性语音障碍， 喉整体张力增加，但无喉痉挛。AdLD的临床结局指标是非 特异于AdLD的主要体征和/或临床上不可行。最近，一种新的自动声学测量， 基于高通基频轮廓（SAHfo）的频谱聚合， 对AdLD特有的LD不连续性敏感。然而，在该措施能够在临床上实施之前， 对于评估SAHfo是否与AdLD的核心体征变化相关和敏感至关重要：内收 喉痉挛和由此产生的非典型发声事件。本提案的目标是调查SAHfo 作为AdLD特异性的临床可行的结局指标。我们将记录高速视频内窥镜检查， 30名成人AdLD患者在最严重的症状状态下的语音声学。在目标1中，我们 确定SAHfo是否反映AdLD的主要体征，将SAHfo（自动计算）与 LD不连续性的数量（从声学信号手动计算）和喉不连续性的数量 痉挛（在高速视频内窥镜检查期间计算）。这一目标的结果将提供必要的证据， SAHfo作为AdLD结局指标的同时有效性。在目标2中，我们将记录来自 30名MTD患者，以评估SAHfo的判别效度，将其在说话者中的值与 AdLD和MTD扬声器。AdLD主要体征的临床可行的客观指标是 对于AdLD患者具有变革性意义，导致早期诊断和成功评估新的 治疗。通过这项工作，申请人将建立在她以前的研究经验与感性和 通过先进声学和高速视频内窥镜方法的培训，的 培训将在波士顿大学进行，并得到她的赞助商Stepp博士和Guenther博士的支持。的 培训计划将包括MATLAB和语音神经控制的正式课程。这项研究将提供 这位有前途的调查员在客观措施的心理测量分析方面拥有经验和专业知识， 她开始了一个成功的职业生涯，作为一个独立的临床科学家在语音障碍。

Project Summary Laryngeal dystonia (LD) is a neurological voice disorder characterized by involuntary spasms of the laryngeal muscles during phonation, which significantly impacts psychosocial well-being and quality of life. The most common subtype of LD is adductor laryngeal dystonia (AdLD), which involves spasms of the laryngeal muscles that adduct the vocal folds, resulting in LD discontinuities. LD discontinuities are disruptions to the speech acoustic signal caused by the spasms and include phonatory breaks (voice breaks) and frequency shifts (pitch breaks) that are most severe during production of voiced sounds. A significant issue in treating AdLD is frequent misdiagnosis. AdLD is often mistaken for muscle tension dysphonia (MTD), a functional voice disorder with increased global laryngeal tension, but without laryngeal spasms. Clinical outcome measures for AdLD are non- specific to the primary signs of AdLD and/or clinically unfeasible . Recently, a new automated acoustic measure, based on spectral aggregate of the high-passed fundamental frequency contour (SAHfo) was designed to be sensitive to LD discontinuities specific to AdLD. However, before the measure can be implemented clinically, it is essential to assess whether SAHfo is related to and sensitive to changes in the core signs of AdLD: adductory laryngeal spasms and the resulting atypical phonatory events. The goal of this proposal is to investigate SAHfo as a clinically feasible outcome measure specific to AdLD. We will record high-speed videoendoscopy and speech acoustics of 30 adults with AdLD while they are in their most symptomatic state. In Aim 1, we will determine whether SAHfo reflects the primary signs of AdLD, comparing SAHfo (calculated automatically) to the number of LD discontinuities (calculated manually from the acoustic signal) and to the number of laryngeal spasms (computed during high-speed videoendoscopy). Results of this aim will provide essential evidence of concurrent validity of SAHfo as an outcome measure in AdLD. In Aim 2, we will record speech acoustics from 30 individuals with MTD to evaluate the discriminant validity of SAHfo, comparing its values in speakers with AdLD and speakers with MTD. A clinically feasible, objective measure of the primary signs of AdLD would be transformative for patients with AdLD, leading to earlier diagnoses and a path to successful evaluation of new treatments. Through this work, the applicant will build on her previous research experience with perceptual and aerodynamic measures by training in advanced acoustics and high-speed videoendoscopic methods. The training will take place at Boston University, with support from her sponsors: Dr. Stepp and Dr. Guenther. The training plan will include formal coursework in MATLAB and neural control of speech. This research will provide this promising investigator with the experience and expertise in psychometric analysis of objective measures as she embarks on a successful career as an independent clinician-scientist in voice disorders.