Digital markers of motor speech impairments in natural speech of patients with ALS-FTD spectrum disorders.
Digital markers of motor speech impairments in natural speech of patients with ALS-FTD spectrum disorders.
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ALS-FTD 谱系障碍患者自然言语中运动言语障碍的数字标记。
DOI：
10.1101/2023.04.29.23289308
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发表时间：
2023
期刊：
影响因子：
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通讯作者：
Nevler,Naomi
中科院分区：
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作者：
Shellikeri,Sanjana;Cho,Sunghye;Ash,Sharon;Gonzalez-Recober,Carmen;McMillan,CoreyT;Elman,Lauren;Quinn,Colin;Amado,DefneA;Baer,Michael;Irwin,DavidJ;Massimo,Lauren;Olm,Chris;Liberman,Mark;Grossman,Murray;Nevler,Naomi
背景与目的ALS-FTSD谱系障碍（ALS-FTSD）患者具有混合的运动和认知障碍，需要有效和定量的评估工具来支持球运动疾病的诊断和跟踪。本研究旨在验证一种新的自动化数字语音工具，该工具可以分析自然连接语音中的元音声学，作为ALS-FTSD患者因球运动疾病导致的发音障碍的标志。方法采用强迫对齐元音自动提取（FAVE）算法，对1分钟录音图片描述进行语音检测和元音声学提取。使用自动声学分析脚本，我们推导了两个发音声学测量：元音空间面积（VSA，在Bark2中）代表舌头的运动范围（大小），元音轨迹的平均第二形成峰斜率（F2斜率）代表舌头的运动速度。我们比较了有和没有临床明显的球运动疾病的ALS （ALS+球与ALS-球）、无运动综合征的行为变异性额颞叶痴呆（bvFTD）和健康对照组（HC）的元音测量。我们将受损的元音测量与球疾病的严重程度相关联，通过临床球评分和感知听者努力程度来评估，并与支配舌头的初级运动皮层口颊部的MRI皮质厚度（oralPMC）相关联。我们还测试了呼吸能力和认知障碍的相关性。结果ALS+球茎45例（男性30例，平均年龄61±11岁），ALS-非球茎22例（男性11例，平均年龄62±10岁），bvFTD 22例（男性13例，平均年龄63±7岁），HC 34例（男性14例，平均年龄69±8岁）。ALS+球的VSA小于ALS-球（VSA: |d|=0.86,p=0.0088； F2斜率：|d|=0.98,p=0.0054）、bvFTD （VSA: |d|=0.67,p=0.043； F2斜率：|d|=1.4,p<0.001）和HC （VSA: |d|=0.73,p=0.024； F2斜率：|d|=1.0,p<0.001）。元音测量随球临床评分的恶化而下降（VSA: R=0.33,p=0.033； F2斜率：R=0.25,p=0.048），较小的VSA与更大的听者努力相关（R=-0.43,p=0.041）。F2斜坡较浅与口腔pmc皮质变薄有关（R=0.50,p=0.03）。元音测量与呼吸和认知测试成绩都没有关系。结论自然语音自动处理提取的元音测量对ALS-FTD患者的球运动疾病敏感，对认知障碍有较强的识别能力。
Background and objectivesPatients with ALS-FTD spectrum disorders (ALS-FTSD) have mixed motor and cognitive impairments and require valid and quantitative assessment tools to support diagnosis and tracking of bulbar motor disease. This study aimed to validate a novel automated digital speech tool that analyzes vowel acoustics from natural, connected speech as a marker for impaired articulation due to bulbar motor disease in ALS-FTSD.MethodsWe used an automatic algorithm called Forced Alignment Vowel Extraction (FAVE) to detect spoken vowels and extract vowel acoustics from 1 minute audio-recorded picture descriptions. Using automated acoustic analysis scripts, we derived two articulatory-acoustic measures: vowel space area (VSA, in Bark2) which represents tongue range-of-motion (size), and average second formant slope of vowel trajectories (F2 slope) which represents tongue movement speed. We compared vowel measures between ALS with and without clinically-evident bulbar motor disease (ALS+bulbar vs. ALS-bulbar), behavioral variant frontotemporal dementia (bvFTD) without a motor syndrome, and healthy controls (HC). We correlated impaired vowel measures with bulbar disease severity, estimated by clinical bulbar scores and perceived listener effort, and with MRI cortical thickness of the orobuccal part of the primary motor cortex innervating the tongue (oralPMC). We also tested correlations with respiratory capacity and cognitive impairment.ResultsParticipants were 45 ALS+bulbar (30 males, mean age=61±11), 22 ALS-nonbulbar (11 males, age=62±10), 22 bvFTD (13 males, age=63±7), and 34 HC (14 males, age=69±8). ALS+bulbar had smaller VSA and shallower average F2 slopes than ALS-bulbar (VSA: |d|=0.86,p=0.0088; F2 slope: |d|=0.98,p=0.0054), bvFTD (VSA: |d|=0.67,p=0.043; F2 slope: |d|=1.4,p<0.001), and HC (VSA: |d|=0.73,p=0.024; F2 slope: |d|=1.0,p<0.001). Vowel measures declined with worsening bulbar clinical scores (VSA: R=0.33,p=0.033; F2 slope: R=0.25,p=0.048), and smaller VSA was associated with greater listener effort (R=-0.43,p=0.041). Shallower F2 slopes were related to cortical thinning in oralPMC (R=0.50,p=0.03). Neither vowel measure was associated with respiratory nor cognitive test scores.ConclusionsVowel measures extracted with automatic processing from natural speech are sensitive to bulbar motor disease in ALS-FTD and are robust to cognitive impairment.