Arnold Shelley S, Payne Jonathan M, McArthur Genevieve, North Kathryn N, Barton Belinda
Kids Neuroscience Centre, The Children's Hospital at Westmead, Sydney, NSW, Australia.
The University of Sydney Children's Hospital Westmead Clinical School, Sydney, NSW, Australia.
J Int Neuropsychol Soc. 2021 May;27(5):484-496. doi: 10.1017/S135561772000106X. Epub 2020 Nov 16.
Reading difficulties are one of the most significant challenges for children with neurofibromatosis type 1 (NF1). The aims of this study were to identify and categorize the types of reading impairments experienced by children with NF1 and to establish predictors of poor reading in this population.
Children aged 7-12 years with NF1 (n = 60) were compared with typically developing children (n = 36). Poor word readers with NF1 were classified according to impairment type (i.e., phonological, surface, mixed), and their reading subskills were compared. A hierarchical multiple regression was conducted to identify predictors of word reading.
Compared to controls, children with NF1 demonstrated significantly poorer literacy abilities. Of the 49 children with NF1 classified as poor readers, 20 (41%) were classified with phonological dyslexia, 24 (49%) with mixed dyslexia, and 5 (10%) fell outside classification categories. Children with mixed dyslexia displayed the most severe reading impairments. Stronger working memory, better receptive language, and fewer inattentive behaviors predicted better word reading skills.
The majority of children with NF1 experience deficits in key reading skills which are essential for them to become successful readers. Weaknesses in working memory, receptive language, and attention are associated with reading difficulties in children with NF1.
阅读困难是1型神经纤维瘤病(NF1)患儿面临的最重大挑战之一。本研究的目的是识别和分类NF1患儿所经历的阅读障碍类型,并确定该人群阅读能力差的预测因素。
将60名7至12岁的NF1患儿与36名发育正常的儿童进行比较。根据障碍类型(即语音、表层、混合型)对NF1的单词阅读能力差的患儿进行分类,并比较他们的阅读子技能。进行分层多元回归以确定单词阅读的预测因素。
与对照组相比,NF1患儿的读写能力明显较差。在49名被归类为阅读能力差的NF1患儿中,20名(41%)被归类为语音性阅读障碍,24名(49%)为混合型阅读障碍,5名(10%)不属于分类范畴。混合型阅读障碍患儿表现出最严重的阅读障碍。更强的工作记忆、更好的接受性语言能力以及较少的注意力不集中行为预示着更好的单词阅读技能。
大多数NF1患儿在关键阅读技能方面存在缺陷,而这些技能是他们成为成功阅读者所必需的。工作记忆、接受性语言和注意力方面的弱点与NF1患儿的阅读困难有关。