1型神经纤维瘤病患者的内化和外化症状：系统评价与荟萃分析
Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis.
作者信息
Liu Dan, Yu Liyan, Wu Xian, Moreira Julia, Mujica Benjamin Felipe, Mukhopadhyay Elora Shelly, Novotney Angelena, Rietman André B, Hou Yang
机构信息
Department of Behavioral Sciences and Social Medicine, College of Medicine, Florida State University, 1115 West Call Street, Tallahassee, FL, 32306-4300, USA.
Department of Biostatistics, College of Public Health, University of Kentucky, Lexington, USA.
出版信息
Syst Rev. 2025 Jan 22;14(1):20. doi: 10.1186/s13643-024-02749-0.
BACKGROUND
Individuals with neurofibromatosis type 1 (NF1) frequently report psychosocial problems, among which internalizing and externalizing symptoms are the most poorly understood due to limited research and inconsistent evidence. This hinders the overall attendance of their psychosocial needs and has a major impact on their quality of life. Thus, this systematic review and meta-analysis was conducted to synthesize existing findings on the degree to which individuals with NF1 experience internalizing and externalizing symptoms, compared with the unaffected population, and explore moderators of the group disparities.
METHODS
Scopus, PsycINFO, Web of Science, PubMed, and ProQuest were searched from inception to March 26th, 2024, which identified 59 eligible studies (N of NF1 = 3182, mean ages 2.38 to 46.4 years). Hedges' g was calculated for differences in internalizing and externalizing symptoms between the NF1 group and the unaffected controls. Study effect sizes were pooled using robust variance estimation and random-effects models. Moderators of group differences were tested using meta-regression.
RESULTS
Random-effects meta-analyses indicated that compared with unaffected controls, individuals with NF1 showed more severe depressive (k = 21; g = 0.43; 95% CI [0.21, 0.65]), anxiety (k = 24; g = 0.27; 95% CI [0.01, 0.54]), somatic (k = 27; g = 0.56; 95% CI [0.30, 0.83]), total internalizing (k = 75; g = 0.50; 95% CI [0.33, 0.67]), aggression (k = 33; g = 0.33; 95% CI [0.08, 0.58]), delinquency, (k = 37; g = 0.43; 95% CI [0.26, 0.60]), and total externalizing symptoms (k = 47; g = 0.24; 95% CI [0.13, 0.35]). Studies that included more participants with NF1 who had ADHD or a lower verbal IQ reported greater group disparities in total internalizing symptoms or aggression.
CONCLUSIONS
Findings highlight the importance of promptly recognizing internalizing and externalizing symptoms in individuals with NF1 for timely interventions. Future research should identify predictors of internalizing and externalizing symptoms within the NF1 population to inform our knowledge and intervention development. Other implications for future research were also discussed.
SYSTEMATIC REVIEW REGISTRATION
The study protocol of this meta-analysis was registered at PROSPERO (CRD42023478258).
背景
1型神经纤维瘤病（NF1）患者经常报告心理社会问题，其中内化和外化症状因研究有限且证据不一致而最不为人所理解。这阻碍了对他们心理社会需求的全面关注，并对他们的生活质量产生重大影响。因此，进行了这项系统评价和荟萃分析，以综合现有研究结果，了解NF1患者与未受影响人群相比内化和外化症状的程度，并探索组间差异的调节因素。
方法
检索了Scopus、PsycINFO、Web of Science、PubMed和ProQuest数据库，时间跨度从创建到2024年3月26日，共识别出59项符合条件的研究（NF1患者n = 3182，平均年龄2.38至46.4岁）。计算了NF1组与未受影响对照组在内化和外化症状方面的差异的Hedges' g值。使用稳健方差估计和随机效应模型对研究效应量进行合并。使用元回归检验组间差异的调节因素。
结果
随机效应荟萃分析表明，与未受影响的对照组相比，NF1患者表现出更严重的抑郁（k = 21；g = 0.43；95% CI [0.21, 0.65]）、焦虑（k = 24；g = 0.27；95% CI [0.01, 0.54]）、躯体症状（k = 27；g = 0.56；95% CI [0.30, 0.83]）、总内化症状（k = 75；g = 0.50；95% CI [0.33, 0.67]）、攻击行为（k = 33；g = 0.33；95% CI [0.08, 0.58]）、违纪行为（k = 37；g = 0.43；95% CI [0.26, 0.60]）和总外化症状（k = 47；g = 0.24；95% CI [0.13, 0.35]）。纳入更多患有注意力缺陷多动障碍（ADHD）或较低语言智商的NF1患者的研究报告了在总内化症状或攻击行为方面更大的组间差异。
结论
研究结果强调了及时识别NF1患者内化和外化症状以便及时干预的重要性。未来的研究应确定NF1人群内化和外化症状的预测因素，以增进我们的认识并推动干预措施的发展。还讨论了对未来研究的其他启示。
系统评价注册
本荟萃分析的研究方案已在PROSPERO注册（CRD42023478258）。
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