Reading skills over time among children with Duchenne muscular dystrophy.

IF 1.6 3区 心理学 Q3 CLINICAL NEUROLOGY Child Neuropsychology Pub Date : 2024-08-01 DOI:10.1080/09297049.2024.2386078
Amanda Kenepp, Shira Russell-Giller, Sonia Seehra, Robert Fee, Veronica J Hinton
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Abstract

Duchenne muscular dystrophy (DMD) is an X-linked progressive neuromuscular disorder with a distinct cognitive profile including decreased verbal span. Children with DMD are also at risk for lower scores on academic achievement tests and increased behavioral problems. Longitudinal analyses generally reveal a stable intellectual profile, although attention and behavioral problems may negatively impact longitudinal IQ scores. To date, no study has reported on reading over time in DMD. Reading performance was assessed longitudinally in children with DMD, examining for potential contributions to the trajectory. Retrospective data analysis on assessments completed at baseline, year 2, and year 4 on 26 boys with DMD and 27 unaffected sibling controls (age at baseline: DMD 8 ± 1.4, controls 9 ± 2.6) indicated that children with DMD performed slightly, yet significantly, worse than controls on reading skills, but the longitudinal trajectory of reading skills for children with DMD and controls was not significantly different. Verbal span at time 1 was uniquely associated with later reading skills in children with DMD. Behavior was not associated with declines. The results confirm that children with DMD underperform on reading tasks and align with previous research suggesting that cognitive skills in DMD are stable over time.

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杜氏肌肉萎缩症儿童的阅读能力随时间的变化。
杜兴氏肌营养不良症(DMD)是一种 X 连锁进行性神经肌肉疾病,具有独特的认知特征,包括语言能力下降。患有 DMD 的儿童还可能在学业成绩测试中得分较低,行为问题增多。尽管注意力和行为问题可能会对纵向智商评分产生负面影响,但纵向分析通常会显示出稳定的智力特征。迄今为止,还没有关于 DMD 患者随时间变化的阅读情况的研究报告。我们对 DMD 患儿的阅读能力进行了纵向评估,以研究其对阅读轨迹的潜在影响。对 26 名 DMD 男孩和 27 名未受影响的对照组兄弟姐妹(基线年龄:DMD 8 ± 1.4,对照组 9 ± 2.6)在基线、第 2 年和第 4 年完成的评估进行的回顾性数据分析显示,DMD 儿童的阅读能力略差于对照组,但差异显著,但 DMD 儿童和对照组儿童的阅读能力纵向轨迹并无显著差异。在第一阶段,DMD 儿童的口头表达能力与后来的阅读能力有着独特的联系。行为与阅读能力的下降无关。研究结果证实,DMD 儿童在阅读任务中表现不佳,这与之前的研究结果一致,表明 DMD 儿童的认知技能随着时间的推移趋于稳定。
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来源期刊
Child Neuropsychology
Child Neuropsychology 医学-临床神经学
CiteScore
4.10
自引率
9.10%
发文量
71
审稿时长
>12 weeks
期刊介绍: The purposes of Child Neuropsychology are to: publish research on the neuropsychological effects of disorders which affect brain functioning in children and adolescents, publish research on the neuropsychological dimensions of development in childhood and adolescence and promote the integration of theory, method and research findings in child/developmental neuropsychology. The primary emphasis of Child Neuropsychology is to publish original empirical research. Theoretical and methodological papers and theoretically relevant case studies are welcome. Critical reviews of topics pertinent to child/developmental neuropsychology are encouraged. Emphases of interest include the following: information processing mechanisms; the impact of injury or disease on neuropsychological functioning; behavioral cognitive and pharmacological approaches to treatment/intervention; psychosocial correlates of neuropsychological dysfunction; definitive normative, reliability, and validity studies of psychometric and other procedures used in the neuropsychological assessment of children and adolescents. Articles on both normal and dysfunctional development that are relevant to the aforementioned dimensions are welcome. Multiple approaches (e.g., basic, applied, clinical) and multiple methodologies (e.g., cross-sectional, longitudinal, experimental, multivariate, correlational) are appropriate. Books, media, and software reviews will be published.
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