Unstuck and on target! for children with neurofibromatosis type 1: a feasibility and acceptability study.

IF 1.9 3区心理学 Q3 CLINICAL NEUROLOGY

Child Neuropsychology Pub Date : 2025-08-27 DOI:10.1080/09297049.2025.2549017

Caitlyn J Cap, Rebecca Levitt, Samantha van Terheyden, Meredith J Goyette, Kaitlyn Tiplady, Karin S Walsh

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Abstract

Children with Neurofibromatosis Type 1 (NF1) experience significant executive dysfunction interfering with outcomes across the lifespan. To date, there have been limited interventions targeting executive function impairments, and even less explored within the NF1 population. The primary purpose of this study was to determine if an existing executive functioning intervention, Unstuck and On Target! (UOT), is a feasible and acceptable intervention for children with NF1. Participants were four children with NF1 (75% male; 75% Caucasian) between the ages of 9 to 12 years and their parents. Participants engaged in 20 weekly group sessions of UOT over the course of five months. Attendance rates and satisfaction ratings were primary outcomes. Attendance was >95% and attrition was null. Parent feedback and satisfaction ratings were overwhelmingly positive. In the first study of its kind, the results suggest that UOT is a feasible and acceptable cognitive intervention for children with NF1. Future development of Phase II/III studies of UOT to examine treatment dose and efficacy is supported.

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脱困，命中目标！儿童1型神经纤维瘤病的可行性和可接受性研究。

患有1型神经纤维瘤病（NF1）的儿童经历了显著的执行功能障碍，影响了整个生命周期的预后。迄今为止，针对执行功能障碍的干预措施有限，在NF1人群中探索的就更少了。本研究的主要目的是确定现有的执行功能干预，Unstuck and On Target！（UOT）是NF1患儿可行且可接受的干预措施。参与者为4名9至12岁的NF1儿童（75%为男性，75%为高加索人）及其父母。在五个月的时间里，参与者每周参加20次UOT小组会议。出勤率和满意度评分是主要结果。出勤率为95%，流失率为零。家长的反馈和满意度评分都非常积极。这是同类研究中的第一项，结果表明，对于NF1儿童，UOT是一种可行且可接受的认知干预。支持未来开展UOT的II/III期研究，以检查治疗剂量和疗效。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Child Neuropsychology 医学-临床神经学

CiteScore

4.10

自引率

9.10%

发文量

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.