Systematic Review and Meta-Analysis: Attention-Deficit/Hyperactivity Disorder Symptoms in Children With Neurofibromatosis Type 1.

IF 9.2 1区医学 Q1 PEDIATRICS

Journal of the American Academy of Child and Adolescent Psychiatry Pub Date : 2024-12-19 DOI:10.1016/j.jaac.2024.09.011

Yang Hou, Liyan Yu, Dan Liu, Emma Wilson-Lemoine, Xian Wu, Julia P Moreira, Benjamin F Mujica, Elora S Mukhopadhyay, Angelena N Novotney, Jonathan M Payne

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引用次数: 0

Abstract

Objective: This meta-analysis aimed to robustly estimate differences in attention-deficit/hyperactivity disorder (ADHD) symptoms between children and adolescents with and without neurofibromatosis type 1 (NF1).

Method: Systematic literature searches were conducted in Scopus, PsycINFO, Web of Science, PubMed, and ProQuest in September 2022, with a supplemental search conducted in Google Scholar in February 2023. The searches identified 2,153 unique articles. Screening identified 114 academic journal articles that assessed parent/caregiver- or teacher-reported ADHD symptoms for children/adolescents with NF1. Two researchers independently screened articles and extracted data. The primary outcome was group differences in ADHD symptoms between children/adolescents with and without NF1 (Hedges g). Data were analyzed using robust variance estimation and random-effects models.

Results: The meta-analysis included 70 studies (138 effect sizes), involving 3,653 children/adolescents with NF1 (46% female; mean age = 9.69 years, SD = 2.60 years) and 4,895 children/adolescents without NF1 (48% female; mean age = 10.03 years, SD = 3.10 years). According to parent/caregiver reports, children/adolescents with NF1 exhibited more severe inattentive symptoms (g = 1.20; 95% CI = 1.06-1.35), hyperactive/impulsive symptoms (g = 0.85; 95% CI = 0. 68-1.03), and combined ADHD symptoms (g = 1.02; 95% CI = 0.87-1.17) than unaffected controls. Inattentive ADHD symptoms were more elevated than hyperactivity/impulsivity for children/adolescents with NF1. Larger effect sizes for inattention and hyperactivity/impulsivity were associated with older age, lower intelligence quotient (IQ), and parent/caregiver vs teacher reports.

Conclusion: NF1 is a monogenic condition that has strong associations with elevated ADHD symptoms. Findings highlight the importance of early intervention and targeted support for ADHD-related problems in children with NF1.

Study preregistration information: Compare the ADHD problems between NF1 and control groups; https://www.crd.york.ac.uk/PROSPERO/display_record.php?RecordID=462063.

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系统评价和荟萃分析：1型神经纤维瘤病儿童的注意缺陷/多动障碍症状

目的：本荟萃分析旨在可靠地估计患有和不患有1型神经纤维瘤病（NF1）的儿童和青少年在注意缺陷/多动障碍（ADHD）症状方面的差异。方法：于2022年9月在Scopus、PsycINFO、Web of Science、PubMed、ProQuest中进行系统文献检索，并于2023年2月在谷歌Scholar中进行补充检索。搜索结果确定了2153篇独特的文章。筛选确定了114篇学术期刊文章，这些文章评估了父母/照顾者或老师报告的NF1儿童/青少年ADHD症状。两位研究者独立筛选文章并提取数据。主要结局是患有和不患有NF1的儿童/青少年ADHD症状的组间差异（Hedges' g）。数据使用稳健方差估计和随机效应模型进行分析。结果：meta分析包括70项研究（138个效应量），涉及3653名患有NF1的儿童/青少年(46%为女性；Mage = 9.69岁，SD = 2.60岁)和4,895名儿童/青少年无NF1(48%为女性；法师= 10.03年，SD = 3.10年)。根据家长/照顾者的报告，患有NF1的儿童/青少年表现出更严重的注意力不集中症状(g = 1.20；95% CI[1.06, 1.35])，多动/冲动症状(g = 0.85；95% ci[0。68, 1.03])，合并ADHD症状(g = 1.02；95% CI[0.87, 1.17])高于未受影响的对照组。儿童/青少年NF1的注意力不集中ADHD症状比多动/冲动更严重。注意力不集中和多动/冲动的影响较大，与年龄较大、智商较低以及父母/照顾者与教师的报告有关。结论：NF1是一种单基因疾病，与ADHD症状升高密切相关。研究结果强调了早期干预和有针对性的支持对NF1儿童adhd相关问题的重要性。

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

Journal of the American Academy of Child and Adolescent Psychiatry 医学-精神病学

CiteScore

21.00

自引率

1.50%

发文量

1383

审稿时长

53 days

期刊介绍： The Journal of the American Academy of Child & Adolescent Psychiatry (JAACAP) is dedicated to advancing the field of child and adolescent psychiatry through the publication of original research and papers of theoretical, scientific, and clinical significance. Our primary focus is on the mental health of children, adolescents, and families. We welcome unpublished manuscripts that explore various perspectives, ranging from genetic, epidemiological, neurobiological, and psychopathological research, to cognitive, behavioral, psychodynamic, and other psychotherapeutic investigations. We also encourage submissions that delve into parent-child, interpersonal, and family research, as well as clinical and empirical studies conducted in inpatient, outpatient, consultation-liaison, and school-based settings. In addition to publishing research, we aim to promote the well-being of children and families by featuring scholarly papers on topics such as health policy, legislation, advocacy, culture, society, and service provision in relation to mental health. At JAACAP, we strive to foster collaboration and dialogue among researchers, clinicians, and policy-makers in order to enhance our understanding and approach to child and adolescent mental health.