成人普瑞德-威利综合症的词汇和阅读技巧

IF 1.8 3区医学 Q2 AUDIOLOGY & SPEECH-LANGUAGE PATHOLOGY

Journal of Communication Disorders Pub Date : 2025-03-01 DOI:10.1016/j.jcomdis.2025.106508

Christelle Robert , Séverine Estival , Virginie Postal , Virginie Laurier , Fabien Mourre , Julie Tricot , Stéphanie Mathey

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引用次数: 0

摘要

普瑞德-威利综合征（PWS）是一种罕见的遗传性疾病，与整体智力障碍有关。虽然研究证明了语言问题，但对阅读知之甚少，阅读是一种与交流有关的关键语言能力。本研究的目的是调查成人PWS患者的词汇和阅读技能。方法共纳入56例PWS患者，其中女性35例，平均年龄33.64岁，年龄范围19 ~ 57岁。采用标准化纸笔测试检测词汇水平（LexTale-FR测试）和阅读能力（Alouette-R测试）。两个计算机化的任务也被用来评估阅读中词汇和语音过程的效率（词汇和语音决策任务，取自ECCLA软件）。分析并比较神经正常的成人和/或儿童的表现。结果PWS成人的词汇量水平较低（与正常成人相比有3 ~ 5个标准差的差异），阅读能力较差（与9岁儿童水平相当），词汇和非词汇语音加工效率较低。结论：成人PWS患者的词汇和阅读能力普遍受损。这些发现可能有助于临床医生更好地了解这些患者的语言能力。

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Vocabulary and reading skills in adults with Prader-Willi syndrome

Introduction

Prader-Willi syndrome (PWS) is a rare genetic condition associated with global intellectual impairment. While research has evidenced speech problems, little is known about reading, which is a critical language ability involved in communication. The aim of the present study was to investigate vocabulary and reading skills in adults with PWS.

Method

A total of 56 individuals (35 females, mean age = 33.64 years, range = 19–57 years) with PWS participated. Standardized paper and pencil tests were used to examine the level of vocabulary (LexTale-FR test) and reading performance (Alouette-R test). Two computerized tasks were also administered to assess the efficiency of lexical and phonological processes in reading (lexical and phonological decision tasks, taken from the ECCLA software). Performance was analyzed and compared with available norms on neurotypical adults and/or children.

Results

The results showed that adults with PWS had a low level of vocabulary (i.e., three to five standard deviations difference compared to neurotypical adults), poor reading skills (i.e., equivalent to the level of nine-year-olds), and less efficient lexical and non-lexical phonological processes.

Conclusion

The present data suggest a global impairment in vocabulary and reading skills in adults with PWS. These findings might help clinicians to better understand the language abilities of these patients.

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来源期刊

Journal of Communication Disorders AUDIOLOGY & SPEECH-LANGUAGE PATHOLOGY-REHABILITATION

CiteScore

3.30

自引率

5.90%

发文量

审稿时长

>12 weeks

期刊介绍： The Journal of Communication Disorders publishes original articles on topics related to disorders of speech, language and hearing. Authors are encouraged to submit reports of experimental or descriptive investigations (research articles), review articles, tutorials or discussion papers, or letters to the editor ("short communications"). Please note that we do not accept case studies unless they conform to the principles of single-subject experimental design. Special issues are published periodically on timely and clinically relevant topics.