Systematic Review and Meta-Analysis: Attention-Deficit/Hyperactivity Disorder Symptoms in Children With Neurofibromatosis Type 1 - 27/03/25
, Liyan Yu, PhD a, Dan Liu, PhD a, Emma Wilson-Lemoine, PhD b, Xian Wu, PhD c, Julia P. Moreira, BS a, Benjamin F. Mujica, BS a, Elora S. Mukhopadhyay, BS a, Angelena N. Novotney, earning BS a, Jonathan M. Payne, DPsych dAbstract |
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; display_record.php?RecordID=462063.
Il testo completo di questo articolo è disponibile in PDF.Key words : attention deficit disorder with hyperactivity, neurofibromatosis type 1, child, meta-analysis, systematic review
Mappa
| This project was funded by a) the Congressionally Directed Medical Research Programs (CDMRP) through the Neurofibromatosis Research Program (NFRP) under Award Number (W81XWH2110504), United States; b) the Florida State University Faculty Startup Funding, United States; and c) the University of Kentucky Faculty Startup Funding, United States. |
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| This study was presented as a poster at the Society for Research in Child Development Biennial Meeting; March 23–25, 2023; Salt Lake City, Utah. The study was also presented as a virtual poster at the Children’s Tumor Foundation annual NF conference; June 24–27, 2023; Scottsdale, Arizona. |
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| This work has been prospectively registered: display_record.php?RecordID=462063. |
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| Data Sharing: Data collected for the study will not be made available to others. No individual-level data were collected. All study-level data meta-analyzed have been provided in the supplemental document. |
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| Drs. Hou and Yu served as the statistical experts for this research. |
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| The authors thank Joel Killam, BS, Lauren Morey, BS, Mary-Mac Chown, BS, Devin Guy, BS, Carson Maun, BS, Denny Oliveira, BS, and Taylor Collins, BS, of Florida State University, for their help with literature search, screening, or data extraction. |
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| Disclosure: Dr. Hou has received funding from the Children’s Tumor Foundation (CTF-2023-10-002) and Florida State University (2024 SEED Grant Award). Dr. Wilson-Lemoine has received funding from the Economic and Social Research Council, Centre for Society and Mental Health at King’s College London [ES/S012567/1] between 2020 – 2023. Dr. Wu has received funding from the National Institute on Aging P01AG078116, the Alzheimer’s Association/the National Alzheimer’s Coordinating Center (NACC) NIAP24-1276268, and the University of Kentucky Center for Clinical and Translational Science (CCTS) DREAM Scholar Program. Dr. Payne has received funding from a Murdoch Children’s Research Institute Clinician Scientist Fellowship. Drs. Yu and Liu, Ms. Moreira, Mr. Mujica, and Mss. Mukhopadhyay and Novotney have reported no biomedical financial interests or potential conflicts of interest. |
Vol 64 - N° 4
P. 447-462 - aprile 2025 Ritorno al numero
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