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PMID: 41989438 已发表 · aheadofprint 英语

Everyday executive functions in children with neurofibromatosis type 1: Data from nine institutions.

Neuropsychology ·2026-04-16

Zong X, Liu D, Wu X, Wolters PL, Payne JM, Klein-Tasman BP, Walsh KS, Janusz J, Pride N, Morris SM, Bearden CE, Stavinoha PL, Hou Y

摘要

Executive function (EF) difficulties are prevalent in neurogenetic syndromes such as Neurofibromatosis Type 1 (NF1), the most common autosomal dominant condition associated with nervous system tumors. However, age-related differences in EF problems in children with NF1 remain poorly understood. This study aimed to examine how EF problems in daily life vary across ages throughout childhood and adolescence and how these age patterns vary across demographic and NF1 disease-related characteristics. This study integrated data of 1,294 observations from 1,025 children with NF1 (Mage = 9.77 years, SD = 4.06, range = 3-18) from nine institutions in the United States and Australia. EF was reported by parents using the Behavior Rating Inventory of Executive Function. Time-varying effect modeling was used to estimate curvilinear EF trends. Children with NF1 demonstrated elevated EF problems compared to the normative mean from early childhood through late adolescence, with deviations generally largest from midchildhood through midadolescence. At ages showing the greatest deviations from the normative mean, medium to large EF gaps were observed across five core domains (Cohen's d = 0.73-1.42). The exact age patterns varied across EF domains and by children's sex, parental education, and NF1 inheritance. Leveraging the largest EF data set of children with NF1 to date, this study precisely estimated EF problems across developmental periods and highlighted the heterogeneity in EF age patterns. These age-specific estimates are crucial for establishing EF developmental norms that guide clinical practice and patient management strategies. The findings can inform the design of individualized intervention and prevention strategies tailored to various developmental periods and diverse demographic and disease-related characteristics for children with NF1. (PsycInfo Database Record (c) 2026 APA, all rights reserved).

文献信息
期刊
Neuropsychology
期刊简称
Neuropsychology
ISSN
1931-1559
发表日期
2026-04-16
语言
英语
国家/地区
United States
NLM ID
8904467
分析服务
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