Written language achievement in children and adolescents with neurofibromatosis type 1 and Plexiform Neurofibromas.

IF 1.6 3区 心理学 Q3 CLINICAL NEUROLOGY Child Neuropsychology Pub Date : 2024-10-01 Epub Date: 2024-02-06 DOI:10.1080/09297049.2024.2307663
Atara Siegel, Mary Anne Toledo-Tamula, Staci Martin, Andy Gillespie, Anne Goodwin, Brigitte Widemann, Pamela L Wolters
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Abstract

Neurofibromatosis type 1 (NF1) is associated with below average writing achievement. However, little is known about specific aspects of written language impacted by NF1, changes in writing over time, and associations between cognitive aspects of the NF1 phenotype and writing. At three timepoints over six years, children with NF1 and plexiform neurofibromas (PNs) completed Woodcock-Johnson tests of writing mechanics (Spelling, Punctuation & Capitalization, handwriting), written expression of ideas (Writing Samples), writing speed (Writing Fluency), and tests of general cognitive ability, executive function, memory, and attention. Children (N = 76, mean age = 12.8 ± 3.4 years) completed at least one baseline writing subtest. Overall writing scores were in the Average range (M = 93.4, SD = 17.4), but lower than population norms (p = 0.002). Scores were highest on Writing Samples (M = 95.2, SD = 17.3), and lowest for Punctuation & Capitalization (M = 87.9, SD = 18.8, p = 0.034). Writing scores were mostly stable over time. Nonverbal reasoning was related to some tests of writing mechanics and written expression of ideas. Short-term memory and inattention explained additional variance in Writing Samples and Spelling. Poor handwriting was associated with writing content beyond the impact of cognitive factors. Children with NF1 and PNs may benefit from early screening and writing support. Interventions should address the contribution of both cognitive and handwriting difficulties in written language.

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患有神经纤维瘤病 1 型和丛状神经纤维瘤的儿童和青少年的书面语言成绩。
神经纤维瘤病 1 型(NF1)与写作成绩低于平均水平有关。然而,人们对 NF1 影响书面语言的具体方面、书写随时间的变化以及 NF1 表型的认知方面与书写之间的关联知之甚少。在六年中的三个时间点,NF1 和丛状神经纤维瘤(PNs)患儿完成了伍德科克-约翰逊(Woodcock-Johnson)写作技巧测试(拼写、标点和大小写、手写)、书面思想表达测试(写作样本)、写作速度测试(写作流畅度)以及一般认知能力、执行功能、记忆力和注意力测试。儿童(人数 = 76,平均年龄 = 12.8 ± 3.4 岁)至少完成了一项基线写作测试。总体写作得分在平均范围内(中=93.4,标差=17.4),但低于人口常模(p=0.002)。写作范文得分最高(中=95.2,标点=17.3),标点和大小写得分最低(中=87.9,标点=18.8,p=0.034)。写作分数随着时间的推移基本保持稳定。非语言推理与某些写作技巧和书面表达想法的测试有关。短期记忆和注意力不集中解释了写作样本和拼写方面的其他差异。除认知因素的影响外,书写不规范还与写作内容有关。患有 NF1 和 PNs 的儿童可能会受益于早期筛查和书写支持。干预措施应同时解决认知障碍和书写障碍对书面语言的影响。
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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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