Adlof Suzanne M, Klusek Jessica, Shinkareva Svetlana V, Robinson Marissa L, Roberts Jane E
Department of Communication Sciences and Disorders, University of South Carolina, Columbia, SC, USA.
J Child Psychol Psychiatry. 2015 Jan;56(1):30-9. doi: 10.1111/jcpp.12267. Epub 2014 Jun 3.
Reading delays are well documented in children with fragile X syndrome (FXS), but few studies have examined linguistic precursors of reading in this population. This study examined the longitudinal development of phonological awareness and its relationship with basic reading in boys with FXS. Individual differences in genetic, social-behavioral and environmental factors were also investigated as predictors of phonological awareness.
Participants included 54 boys with FXS and 53 typically developing (TD) mental age-matched peers who completed assessments of phonological awareness, nonverbal intelligence, and reading annually for up to 4 years. FMRP level and autism symptomatology were also measured within the FXS group. Hierarchical linear modeling was used to examine change in phonological awareness over time and its predictors. Linear regression was used to examine phonological awareness as a predictor of word reading.
Boys with FXS exhibited slower growth than TD peers in phonological awareness only when nonverbal cognitive abilities were not controlled. The rate of change in phonological awareness decreased significantly after age 10 in boys with FXS. Phonological awareness accounted for 18% unique variance in basic reading ability after controlling for nonverbal cognition, with similar relationships across groups.
Phonological awareness skills in the boys with FXS were commensurate with their nonverbal cognitive abilities, with similar relationships between phonological awareness and reading as observed in the TD mental age-matched peers. More research is needed to examine potential causal relationships between phonological awareness, other language skills, and reading abilities in individuals with FXS and other neurodevelopmental disorders.
脆性X综合征(FXS)患儿的阅读延迟现象已有充分记录,但很少有研究考察该群体阅读的语言先兆。本研究考察了FXS男孩语音意识的纵向发展及其与基础阅读的关系。还调查了遗传、社会行为和环境因素的个体差异作为语音意识的预测因素。
参与者包括54名患有FXS的男孩和53名发育正常(TD)且心理年龄匹配的同龄人,他们每年完成语音意识、非言语智力和阅读评估,为期4年。FXS组还测量了FMRP水平和自闭症症状。使用分层线性模型来考察语音意识随时间的变化及其预测因素。使用线性回归来考察语音意识作为单词阅读的预测因素。
仅在未控制非言语认知能力时,FXS男孩在语音意识方面的增长比TD同龄人慢。FXS男孩在10岁后语音意识的变化率显著下降。在控制非言语认知后,语音意识在基础阅读能力中占18%的独特方差,各群体之间的关系相似。
FXS男孩的语音意识技能与其非言语认知能力相当,语音意识与阅读之间的关系与TD心理年龄匹配的同龄人相似。需要更多研究来考察FXS和其他神经发育障碍个体中语音意识、其他语言技能和阅读能力之间的潜在因果关系。