Komesidou Rouzana, Brady Nancy C, Fleming Kandace, Esplund Amy, Warren Steven F
University of Kansas, Lawrence.
J Speech Lang Hear Res. 2017 Feb 1;60(2):422-434. doi: 10.1044/2016_JSLHR-L-15-0360.
This research explored syntactic growth in children with fragile X syndrome (FXS) over a 5-year period, and variability in growth in relation to autism symptoms, nonverbal cognition, maternal responsivity, and gender.
Language samples at 4 time points from 39 children with FXS, 31 boys and 8 girls, were analyzed using the Index of Productive Syntax (Scarborough, 1990) and mean length of utterance (Brown, 1973). The degree of autism symptoms was evaluated using the Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) at the first time point. Maternal responsivity estimates were averaged across time points.
Children with FXS showed significant syntactic growth over time and a significant plateau (quadratic trend) in the later observations. Children who exhibited more autism symptoms at Time 1 had significantly lower syntactic abilities over time than children who exhibited fewer autism symptoms. Nonverbal cognition significantly predicted mean length of utterance scores but not Index of Productive Syntax scores. Maternal responsivity was not a significant predictor of syntactic outcomes. Girls with FXS generally demonstrated better expressive syntax than boys with FXS with notable individual differences.
Despite significant growth over time, expressive syntax is a vulnerable domain for children with FXS, especially for those with severe autism symptoms. Clinical implications arising from the current findings are discussed.
本研究探讨了脆性X综合征（FXS）患儿在5年期间的句法发展，以及与自闭症症状、非语言认知、母亲反应性和性别的发展差异。
使用句法产出指数（斯卡伯勒，1990年）和话语平均长度（布朗，1973年），对39名FXS患儿（31名男孩和8名女孩）在4个时间点的语言样本进行分析。在第一个时间点，使用儿童自闭症评定量表（肖普勒、赖克勒和伦纳，1988年）评估自闭症症状的程度。对各时间点的母亲反应性估计值进行平均。
FXS患儿的句法能力随时间显著发展，且在后期观察中出现显著的平稳期（二次趋势）。在时间1表现出更多自闭症症状的儿童，其随时间推移的句法能力显著低于自闭症症状较少的儿童。非语言认知显著预测了话语平均长度得分，但不能预测句法产出指数得分。母亲反应性不是句法结果的显著预测因素。FXS女孩通常比FXS男孩表现出更好的表达性句法，且存在显著的个体差异。
尽管随时间有显著发展，但表达性句法对于FXS患儿来说是一个易受影响的领域，尤其是对于那些有严重自闭症症状的患儿。讨论了当前研究结果的临床意义。
