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系统评价与荟萃分析:1型神经纤维瘤病患儿的注意力缺陷/多动障碍症状

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

作者信息

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

机构信息

Florida State University, Tallahassee, Florida, USA.

Florida State University, Tallahassee, Florida, USA.

出版信息

J Am Acad Child Adolesc Psychiatry. 2025 Apr;64(4):447-462. doi: 10.1016/j.jaac.2024.09.011. Epub 2024 Dec 19.

DOI:10.1016/j.jaac.2024.09.011
PMID:39709008
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.

PLAIN LANGUAGE SUMMARY

Increasing evidence has suggested a higher risk for attention-deficit/hyperactivity disorder (ADHD) in individuals with neurofibromatosis type 1 (NF1). In this study of ADHD symptom severity in youth with NF1, the authors analyzed data from 70 articles. The authors found much more severe ADHD symptoms in children and adolescents with NF1 compared to youth without NF1. Inattentive symptoms were more pronounced in children with NIF who were older or had a lower IQ.

STUDY PREREGISTRATION INFORMATION

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

摘要

目的

本荟萃分析旨在可靠地估计1型神经纤维瘤病(NF1)患儿及青少年与无NF1的患儿及青少年在注意力缺陷多动障碍(ADHD)症状方面的差异。

方法

2022年9月在Scopus、PsycINFO、Web of Science、PubMed和ProQuest中进行了系统文献检索,并于2023年2月在谷歌学术中进行了补充检索。检索共识别出2153篇独特文章。筛选出114篇学术期刊文章,这些文章评估了NF1患儿/青少年的父母/照料者或教师报告的ADHD症状。两名研究人员独立筛选文章并提取数据。主要结局是有和无NF1的患儿/青少年在ADHD症状方面的组间差异(Hedges g)。使用稳健方差估计和随机效应模型对数据进行分析。

结果

该荟萃分析纳入了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;平均年龄=9.69岁,标准差=2.60岁)和4895名无NF1的儿童/青少年(48%为女性;平均年龄=10.03岁,标准差=3.10岁)。根据父母/照料者报告,与未受影响的对照组相比,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%置信区间=1.06 - 1.35)、多动/冲动症状(g=0.85;95%置信区间=0.68 - 1.03)以及合并的ADHD症状(g=1.02;95%置信区间=0.87 - 1.17)。对于患有NF1的儿童/青少年,注意力不集中的ADHD症状比多动/冲动症状更为突出。注意力不集中和多动/冲动的效应量较大与年龄较大、智商较低以及父母/照料者与教师报告有关。

结论

NF1是一种单基因疾病,与ADHD症状升高密切相关。研究结果凸显了对NF1患儿ADHD相关问题进行早期干预和针对性支持的重要性。

通俗易懂的总结

越来越多的证据表明,1型神经纤维瘤病(NF1)患者患注意力缺陷多动障碍(ADHD)的风险更高。在这项关于NF1青少年ADHD症状严重程度的研究中,作者分析了70篇文章的数据。作者发现,与无NF1的青少年相比,患有NF1的儿童和青少年的ADHD症状要严重得多。在年龄较大或智商较低的NF1患儿中,注意力不集中症状更为明显。

研究预注册信息

比较NF1组和对照组之间的ADHD问题;https://www.crd.york.ac.uk/PROSPERO/display_record.php?RecordID=462063

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