Language development in FXS (Brady et al., 2020)

Purpose: The aim of this study was to investigate language growth in individuals with fragile X syndrome (FXS) from early childhood to adolescence and the influence of maternal responsivity on language growth.Method: We conducted a longitudinal analysis of language development in 55 youths (44 males, 11 females) with FXS. Data collection spanned the ages of 11–216 months. We measured expressive and receptive vocabulary with standardized tests. The number of different words and mean length of utterance were obtained from language sample analyses of mother–child interactions. We also measured maternal comments (responsivity indicator) produced during the language samples and child nonverbal IQ.Results: Growth models indicated that rates of number of different words and receptive vocabulary were related to maternal commenting. Mean length of utterance did not change significantly over time. Expressive vocabulary measured with a standardized test grew, but the growth was not related to maternal commenting. Nonverbal IQ was related to all language outcomes at age of 10 years and to changes over time in vocabulary. Visual analysis indicated that the highest scores on standardized tests were produced by girls; however, measures derived from language sample analyses appeared similar for boys and girls. Language models for boys only were similar to the total sample models with lower scores at age of 10 years for some outcomes.Conclusion: Results of persistent language impairments for most youths with FXS suggest the need for continued, focused interventions aimed at improved language productions in addition to a responsive environment.Supplemental Material S1. Models rerun excluding females. Brady, N. C., Fleming, K., Bredin-Oja, S. L., Fielding-Gebhardt, H., & Warren, S. F. (2020). Language development from early childhood to adolescence in youths with fragile X syndrome. Journal of Speech, Language, and Hearing Research. Advance online publication. https://doi.org/10.1044/2020_JSLHR-20-00198

研究目的：本研究旨在探讨脆性X综合征（FXS）个体从幼儿期至青春期的语言发展，以及母性反应性对语言发展的影响。研究方法：我们对55名脆性X综合征青年（男性44名，女性11名）的语言发展进行了纵向分析。数据收集时间跨度为11至216个月。通过标准化测试测量了表达性和接受性词汇。从母亲与儿童的互动语言样本分析中获得了不同单词的数量和平均话语长度。我们还测量了在语言样本期间产生的母性评论（反应性指标）以及儿童的非言语智商。研究结果：增长模型表明，不同单词的数量和接受性词汇的增长率与母性评论相关。平均话语长度在时间上没有显著变化。通过标准化测试测量的表达性词汇有所增长，但增长与母性评论无关。非言语智商在10岁时与所有语言结果相关，并且与词汇随时间的变化相关。视觉分析表明，标准化测试的最高分数由女孩产生；然而，从语言样本分析中得出的测量值在男孩和女孩之间相似。仅针对男孩的语言模型与总体样本模型相似，但在10岁时某些结果得分较低。研究结论：大多数脆性X综合征青年持续存在的语言障碍结果表明，除了响应性环境外，还需要持续、有针对性的干预措施，旨在提高语言产出。补充材料S1：排除女性的重新运行模型。Brady, N. C., Fleming, K., Bredin-Oja, S. L., Fielding-Gebhardt, H., & Warren, S. F. (2020). 脆性X综合征个体从幼儿期至青春期的语言发展。言语、语言和听力研究杂志。在线预发表。https://doi.org/10.1044/2020_JSLHR-20-00198