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Predictors of cognitive development in children with neurofibromatosis type 1 and plexiform neurofibromas.


ABSTRACT:

Aim

To describe the cognitive development of children with neurofibromatosis type 1 (NF1) and plexiform neurofibromas, and identify predictors of cognitive development.

Method

Participants included 88 children with NF1 and plexiform neurofibromas (50 males, 38 females, aged 6-18y, mean=12y, SD=3y 7mo) on a natural history study at the National Cancer Institute. Neuropsychological assessments (e.g. IQ, academic achievement, attention, and executive functioning) were administered three times over 6 years.

Results

Relative to normative peers, the total sample of children with NF1 and plexiform neurofibromas demonstrated significantly lower scores in most cognitive domains and decreasing z-scores over time in math, writing, inhibitory control, and working memory. Children who had parents with (vs without) NF1 were more likely to experience decreased z-scores in performance IQ, reading, writing, attention, and working memory. Higher (vs lower) parental education was related to higher levels of IQ, math, reading, and cognitive flexibility and a slower decrease in math z-scores. Children's sex and the number of NF1 disease-related complications were not related to most cognitive outcomes.

Interpretation

Children with NF1 and plexiform neurofibromas are at high risk for cognitive difficulties and declining z-scores in various domains of cognitive functioning over time. The findings highlight the need for a better understanding of the within-group differences in these children and their need for individualized educational plans.

What this paper adds

Math, writing, inhibitory control, and working memory scores decreased over time. The proportion of children with clinically significant cognitive deficits increased over time. Parental neurofibromatosis type 1 and low education were related to greater cognitive difficulties in children.

SUBMITTER: Hou Y 

PROVIDER: S-EPMC7332409 | biostudies-literature | 2020 Aug

REPOSITORIES: biostudies-literature

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Publications

Predictors of cognitive development in children with neurofibromatosis type 1 and plexiform neurofibromas.

Hou Yang Y   Allen Taryn T   Wolters Pamela L PL   Toledo-Tamula Mary Anne MA   Martin Staci S   Baldwin Andrea A   Reda Stephanie S   Gillespie Andy A   Goodwin Anne A   Widemann Brigitte C BC  

Developmental medicine and child neurology 20200212 8


<h4>Aim</h4>To describe the cognitive development of children with neurofibromatosis type 1 (NF1) and plexiform neurofibromas, and identify predictors of cognitive development.<h4>Method</h4>Participants included 88 children with NF1 and plexiform neurofibromas (50 males, 38 females, aged 6-18y, mean=12y, SD=3y 7mo) on a natural history study at the National Cancer Institute. Neuropsychological assessments (e.g. IQ, academic achievement, attention, and executive functioning) were administered th  ...[more]

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