Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis
Abstract 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 psychosocia...
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2025-01-01
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| Online Access: | https://doi.org/10.1186/s13643-024-02749-0 |
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| author | Dan Liu Liyan Yu Xian Wu Julia Moreira Benjamin Felipe Mujica Elora Shelly Mukhopadhyay Angelena Novotney André B. Rietman Yang Hou |
| author_facet | Dan Liu Liyan Yu Xian Wu Julia Moreira Benjamin Felipe Mujica Elora Shelly Mukhopadhyay Angelena Novotney André B. Rietman Yang Hou |
| author_sort | Dan Liu |
| collection | DOAJ |
| description | Abstract 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). |
| format | Article |
| id | doaj-art-746beacda3d94d8daa078ac36fb1e4d7 |
| institution | Kabale University |
| issn | 2046-4053 |
| language | English |
| publishDate | 2025-01-01 |
| publisher | BMC |
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| spelling | doaj-art-746beacda3d94d8daa078ac36fb1e4d72025-01-26T12:17:34ZengBMCSystematic Reviews2046-40532025-01-0114111410.1186/s13643-024-02749-0Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysisDan Liu0Liyan Yu1Xian Wu2Julia Moreira3Benjamin Felipe Mujica4Elora Shelly Mukhopadhyay5Angelena Novotney6André B. Rietman7Yang Hou8Department of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Biostatistics, College of Public Health, University of KentuckyDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityDepartment of Child and Adolescent Psychiatry/Psychology, Erasmus Medical Centre - Sophia Children’s HospitalDepartment of Behavioral Sciences and Social Medicine, College of Medicine, Florida State UniversityAbstract 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).https://doi.org/10.1186/s13643-024-02749-0Systematic reviewMeta-analysisNeurofibromatosisInternalizing problemsExternalizing problemsPredictors |
| spellingShingle | Dan Liu Liyan Yu Xian Wu Julia Moreira Benjamin Felipe Mujica Elora Shelly Mukhopadhyay Angelena Novotney André B. Rietman Yang Hou Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis Systematic Reviews Systematic review Meta-analysis Neurofibromatosis Internalizing problems Externalizing problems Predictors |
| title | Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis |
| title_full | Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis |
| title_fullStr | Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis |
| title_full_unstemmed | Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis |
| title_short | Internalizing and externalizing symptoms in individuals with neurofibromatosis type 1: a systematic review and meta-analysis |
| title_sort | internalizing and externalizing symptoms in individuals with neurofibromatosis type 1 a systematic review and meta analysis |
| topic | Systematic review Meta-analysis Neurofibromatosis Internalizing problems Externalizing problems Predictors |
| url | https://doi.org/10.1186/s13643-024-02749-0 |
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