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本文引用的文献

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Longitudinal profiles of expressive vocabulary, syntax and pragmatic language in boys with fragile X syndrome or Down syndrome.脆性 X 综合征或唐氏综合征男孩表达性词汇、句法和语用语言的纵向特征。
Int J Lang Commun Disord. 2013 Jul-Aug;48(4):432-43. doi: 10.1111/1460-6984.12019. Epub 2013 Apr 19.
2
Learning to read in Williams syndrome and Down syndrome: syndrome-specific precursors and developmental trajectories.学习阅读在威廉姆斯综合征和唐氏综合征中的表现:综合征特异性的前期征兆和发展轨迹。
J Child Psychol Psychiatry. 2013 Jul;54(7):754-62. doi: 10.1111/jcpp.12070. Epub 2013 May 29.
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Social communication and theory of mind in boys with autism and fragile x syndrome.患有自闭症和脆性X综合征男孩的社交沟通与心理理论
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The growth of reading skills in children with Down Syndrome.唐氏综合征儿童阅读技能的发展。
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Language Development in Individuals with Fragile X Syndrome.脆性X综合征患者的语言发展
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Maternal responsivity predicts language development in young children with fragile X syndrome.母亲反应能力可预测脆性 X 综合征幼儿的语言发展。
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Phonological awareness of children with Down syndrome: its role in learning to read and the effectiveness of related interventions.唐氏综合征儿童的语音意识:在阅读学习中的作用及相关干预措施的效果。
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Functional skills of individuals with fragile x syndrome: a lifespan cross-sectional analysis.脆性X综合征患者的功能技能:一项全生命周期横断面分析。
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The fragile X prevalence paradox.脆性X综合征患病率悖论。
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脆性X综合征男孩的语音意识与阅读

Phonological awareness and reading in boys with fragile X syndrome.

作者信息

Adlof Suzanne M, Klusek Jessica, Shinkareva Svetlana V, Robinson Marissa L, Roberts Jane E

机构信息

Department of Communication Sciences and Disorders, University of South Carolina, Columbia, SC, USA.

出版信息

J Child Psychol Psychiatry. 2015 Jan;56(1):30-9. doi: 10.1111/jcpp.12267. Epub 2014 Jun 3.

DOI:10.1111/jcpp.12267
PMID:24889646
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4254899/
Abstract

BACKGROUND

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.

METHODS

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.

RESULTS

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.

CONCLUSION

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.

摘要

背景

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。

方法

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。

结果

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。

结论

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。