脆性X综合征男孩的阅读和语音技能
Reading and phonological skills in boys with fragile X syndrome.
作者信息
Klusek Jessica, Hunt Anna W, Mirrett Penny L, Hatton Deborah D, Hooper Stephen R, Roberts Jane E, Bailey Donald B
机构信息
Department of Psychology, University of South Carolina, Barnwell College, 1512 Pendleton Street, Columbia, SC, 29208, USA,
出版信息
J Autism Dev Disord. 2015 Jun;45(6):1699-711. doi: 10.1007/s10803-014-2328-y.
Abstract
Although reading skills are critical for the success of individuals with intellectual disabilities, literacy has received little attention in fragile X syndrome (FXS). This study examined the literacy profile of FXS. Boys with FXS (n = 51; mean age 10.2 years) and mental age-matched boys with typical development (n = 35) participated in standardized assessments of reading and phonological skills. Phonological skills were impaired in FXS, while reading was on-par with that of controls. Phonological awareness predicted reading ability and ASD severity predicted poorer phonological abilities in FXS. Boys with FXS are capable of attaining reading skills that are commensurate with developmental level and phonological awareness skills may play a critical role in reading achievement in FXS.
摘要
尽管阅读技能对于智障人士的成功至关重要，但在脆性X综合征（FXS）中，读写能力却很少受到关注。本研究调查了FXS的读写能力概况。患有FXS的男孩（n = 51；平均年龄10.2岁）和心理年龄匹配的发育正常男孩（n = 35）参与了阅读和语音技能的标准化评估。FXS患者的语音技能受损，而阅读能力与对照组相当。语音意识可预测阅读能力，而自闭症谱系障碍（ASD）严重程度可预测FXS患者较差的语音能力。患有FXS的男孩能够获得与发育水平相称的阅读技能，并且语音意识技能可能在FXS的阅读成绩中起关键作用。
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本文引用的文献
1
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J Child Psychol Psychiatry. 2015 Jan;56(1):30-9. doi: 10.1111/jcpp.12267. Epub 2014 Jun 3.
2
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J Speech Lang Hear Res. 2014 Oct;57(5):1692-707. doi: 10.1044/2014_JSLHR-L-13-0064.
3
Consistency between research and clinical diagnoses of autism among boys and girls with fragile X syndrome.
J Intellect Disabil Res. 2014 Oct;58(10):940-52. doi: 10.1111/jir.12121. Epub 2014 Feb 17.
4
Social communication and theory of mind in boys with autism and fragile x syndrome.
Front Psychol. 2012 Aug 20;3:266. doi: 10.3389/fpsyg.2012.00266. eCollection 2012.
5
Contributions of phonological and verbal working memory to language development in adolescents with fragile X syndrome.
J Neurodev Disord. 2011 Dec;3(4):335-47. doi: 10.1007/s11689-011-9095-2. Epub 2011 Oct 13.
6
Prevalence, phenomenology, aetiology and predictors of challenging behaviour in Smith-Magenis syndrome.
J Intellect Disabil Res. 2011 Feb;55(2):138-51. doi: 10.1111/j.1365-2788.2010.01371.x. Epub 2011 Jan 4.
7
Working memory subsystems and task complexity in young boys with Fragile X syndrome.
J Intellect Disabil Res. 2011 Jan;55(1):19-29. doi: 10.1111/j.1365-2788.2010.01343.x. Epub 2010 Dec 1.
8
Phonological awareness of children with Down syndrome: its role in learning to read and the effectiveness of related interventions.
Res Dev Disabil. 2010 Mar-Apr;31(2):316-30. doi: 10.1016/j.ridd.2009.11.002. Epub 2009 Nov 30.
9
A Comprehensive Profile of Decoding and Comprehension in Autism Spectrum Disorders.
J Autism Dev Disord. 2010 Apr;40(4):485-93. doi: 10.1007/s10803-009-0892-3.
10
Reading and arithmetic in adolescents with autism spectrum disorders: peaks and dips in attainment.
Neuropsychology. 2009 Nov;23(6):718-28. doi: 10.1037/a0016360.
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