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基于模型的自闭症谱系障碍儿童言语障碍病例研究

A Modeling-Guided Case Study of Disordered Speech in Minimally Verbal Children With Autism Spectrum Disorder.

机构信息

MGH Institute of Health Professions, Boston, MA.

Harvard Medical School, Boston, MA.

出版信息

Am J Speech Lang Pathol. 2021 Jun 18;30(3S):1542-1557. doi: 10.1044/2021_AJSLP-20-00121. Epub 2021 Apr 14.

Abstract

Purpose Understanding what limits speech development in minimally verbal (MV) children with autism spectrum disorder (ASD) is important for providing highly effective targeted therapies. This preliminary investigation explores the extent to which developmental speech deficits predicted by Directions Into Velocities of Articulators (DIVA), a computational model of speech production, exemplify real phenotypes. Method Implementing a motor speech disorder in DIVA predicted that speech would become highly variable within and between tokens, while implementing a motor speech plus an auditory processing disorder predicted that DIVA's speech would become highly centralized (schwa-like). Acoustic analyses of DIVA's output predicted that acoustically measured phoneme distortion would be similar between the two cases, but that in the former case, speech would show more within- and between-token variability than in the latter case. We tested these predictions quantitatively on the speech of children with MV ASD. In Study 1, we tested the qualitative predictions using perceptual analysis methods. Speech pathologists blinded to the purpose of the study tallied the signs of childhood apraxia of speech that appeared in the speech of 38 MV children with ASD. K-means clustering was used to create two clusters from the group of 38, and analysis of variance was used to determine whether the clusters differed according to perceptual features corresponding to within- and between-token variability. In Study 2, we employed acoustic analyses on the speech of the child from each cluster who produced the largest number of analyzable tokens to test the predictions of differences in within-token variability, between-token variability, and vowel space area. Results Clusters produced by k-means analysis differed by perceptual features that corresponded to within-token variability. Nonsignificant differences between clusters were found for features corresponding to between-token variability. Subsequent acoustic analyses of the selected cases revealed that the speech of the child from the high-variability cluster showed significantly more quantitative within- and between-token variability than the speech of the child from the low-variability cluster. The vowel space of the child from the low-variability cluster was more centralized than that of typical children and that of the child from the high-variability cluster. Conclusions Results provide preliminary evidence that subphenotypes of children with MV ASD may exist, characterized by (a) comorbid motor speech disorder and (b) comorbid motor speech plus auditory processing disorder. The results motivate testable predictions about how these comorbidities affect speech. Supplemental Material https://doi.org/10.23641/asha.14384432.

摘要

目的 理解自闭症谱系障碍(ASD)中轻度言语(MV)儿童的言语发展受限的原因,对于提供高效的靶向治疗非常重要。本初步研究旨在探讨语音产生的计算模型 Directions Into Velocities of Articulators(DIVA)预测的发育性言语缺陷在多大程度上能体现真实的表型。 方法 在 DIVA 中实施运动性言语障碍的预测结果表明,言语在音位内和音位间的变异性会很高,而在 DIVA 中同时实施运动性言语和听觉处理障碍的预测结果表明,DIVA 的言语会变得高度集中(类“schwa”)。对 DIVA 输出的声学分析预测,在这两种情况下,通过声学测量得到的音位失真会相似,但在前一种情况下,言语的音位内和音位间的变异性会大于后一种情况。我们在 MV ASD 儿童的言语上定量测试了这些预测。在研究 1 中,我们使用感知分析方法测试了定性预测。对研究目的一无所知的言语病理学家对 38 名患有 ASD 的 MV 儿童的言语进行了言语运动障碍的分类。使用 K 均值聚类从 38 名儿童中创建了两个聚类,并使用方差分析来确定聚类是否根据与音位内和音位间变异性相对应的感知特征而有所不同。在研究 2 中,我们对每个聚类中产生可分析音位最多的儿童的言语进行了声学分析,以测试音位内变异性、音位间变异性和元音空间面积差异的预测。 结果 K 均值分析产生的聚类通过与音位内变异性相对应的感知特征而有所不同。在与音位间变异性相对应的特征上,聚类之间没有显著差异。对所选病例的后续声学分析表明,高变异性聚类中儿童的言语在音位内和音位间的变异性上明显多于低变异性聚类中儿童的言语。低变异性聚类中儿童的元音空间比典型儿童和高变异性聚类中儿童的元音空间更集中。 结论 研究结果初步表明,MV ASD 儿童可能存在亚表型,表现为(a)伴运动性言语障碍和(b)伴运动性言语和听觉处理障碍。这些结果为这些共病如何影响言语提供了可检验的预测。 补充材料 https://doi.org/10.23641/asha.14384432.

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