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典型和非典型的神经机制支持 Angelman 综合征中的口语处理。

Typical and atypical neural mechanisms support spoken word processing in Angelman syndrome.

机构信息

Vanderbilt Kennedy Center for Research on Human Development, Nashville, TN 37203, USA; Vanderbilt University Medical Center, Nashville, TN 37232, USA.

Vanderbilt University, Nashville, TN 37235, USA.

出版信息

Brain Lang. 2023 Jan;236:105215. doi: 10.1016/j.bandl.2022.105215. Epub 2022 Dec 8.

Abstract

Angelman syndrome (AS) is known to affect expressive and receptive communication abilities. This study examined individual differences in neural mechanisms underlying speech processing in children with AS (n = 24, M age = 10.01 years) and typical development (n = 30, M age = 10.82 years) using auditory event-related potentials during passive listening to common English words and novel pseudowords. A group of adults with AS (n = 7, M = 31.78 years) provided data about the upper developmental range. The typically developing group demonstrated the expected more negative amplitudes in response to words than pseudowords within 250-500 ms after stimulus onset at the left temporal scalp region. Children and adults with AS exhibited a similar left-lateralized pattern of word-pseudoword differentiation at temporal and parietal regions, but not the midline parietal memory response for known words observed in the typically developing group, suggesting typical-like word-pseudoword differentiation along with possible alterations in the automatic recall of word meaning. These results have important implications for understanding receptive and expressive communication processes in AS and support the use of auditory neural responses for characterizing individual differences in neurodevelopmental disorders with limited speech.

摘要

天使综合征(AS)已知会影响表达和接受性沟通能力。本研究使用听觉事件相关电位,在被动聆听常见英语单词和新造假词时,检查了 AS 儿童(n=24,M 年龄=10.01 岁)和典型发育儿童(n=30,M 年龄=10.82 岁)的言语处理的神经机制的个体差异。一组患有 AS 的成年人(n=7,M=31.78 岁)提供了关于较高发育范围的数据。与假词相比,典型发育组在刺激后 250-500 毫秒内在左颞区显示出对单词的期望的更负幅度。AS 儿童和成人在颞区和顶区表现出类似的左侧词-假词分化模式,但在典型发育组中没有观察到中线顶区对已知单词的记忆反应,这表明具有可能改变的单词意义自动回忆的类似单词-假词分化。这些结果对理解 AS 中的接受和表达性沟通过程具有重要意义,并支持使用听觉神经反应来描述言语有限的神经发育障碍的个体差异。

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本文引用的文献

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Developmental Skills of Individuals with Angelman Syndrome Assessed Using the Bayley-III.
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Social-emotional processing in nonverbal individuals with Angelman syndrome: evidence from brain responses to known and novel names.
J Intellect Disabil Res. 2019 Mar;63(3):244-254. doi: 10.1111/jir.12570. Epub 2018 Nov 23.
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Exploring autism symptoms in an Australian cohort of patients with Prader-Willi and Angelman syndromes.
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