Johannes Boettcher , Stian Orm , Krister Westlye Fjermestad
{"title":"挪威罕见遗传疾病青少年队列中的自闭症特征、社交退缩以及行为和情绪问题","authors":"Johannes Boettcher , Stian Orm , Krister Westlye Fjermestad","doi":"10.1016/j.ridd.2024.104699","DOIUrl":null,"url":null,"abstract":"<div><h3>Background</h3><p>Adolescents with rare genetic disorders represent a population that can be at risk in psychosocial terms. Despite its importance, the association of autism traits and social withdrawal in behavioral and emotional problems in adolescents with rare genetic disorders remains understudied.</p></div><div><h3>Aim</h3><p>The study aimed to empirically examine the clinical characteristics of adolescents with rare genetic disorders with a behavior theory-driven approach.</p></div><div><h3>Method</h3><p>We investigated the behavioral and emotional problems and current and lifetime autistic traits in a sample of 93 Norwegian adolescents (<em>M</em><sub>age</sub> = 13.2 years, SD<sub>age</sub> = 2.4, range<sub>age</sub> 10–17, 62.4% females, 37.6% males) with various rare genetic disorders. The adolescents were investigated cross-sectionally utilizing standardized psychometric questionnaires rated by their parents.</p></div><div><h3>Results</h3><p>More current and lifetime autistic traits and social withdrawal were all associated with more internalizing problems. Further analyses demonstrated that social withdrawal partially mediated the positive association between current autistic traits and internalizing problems. In contrast, social withdrawal fully mediated the positive association between lifetime autistic traits and internalizing problems.</p></div><div><h3>Conclusion and implications</h3><p>Our results demonstrate important characteristics of adolescents with rare genetic disorders that may guide clinicians and future interventions. Social withdrawal may be prodromal to internalizing problems such as anxiety and depression. Thus, clinically addressing social withdrawal can represent a means to prevent internalizing problems in adolescents with rare genetic disorders and autistic traits.</p></div>","PeriodicalId":51351,"journal":{"name":"Research in Developmental Disabilities","volume":null,"pages":null},"PeriodicalIF":2.9000,"publicationDate":"2024-02-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S0891422224000313/pdfft?md5=37aa672a60cdce910355527aeffbc81a&pid=1-s2.0-S0891422224000313-main.pdf","citationCount":"0","resultStr":"{\"title\":\"Autism traits, social withdrawal, and behavioral and emotional problems in a Norwegian cohort of adolescents with rare genetic disorders\",\"authors\":\"Johannes Boettcher , Stian Orm , Krister Westlye Fjermestad\",\"doi\":\"10.1016/j.ridd.2024.104699\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Background</h3><p>Adolescents with rare genetic disorders represent a population that can be at risk in psychosocial terms. Despite its importance, the association of autism traits and social withdrawal in behavioral and emotional problems in adolescents with rare genetic disorders remains understudied.</p></div><div><h3>Aim</h3><p>The study aimed to empirically examine the clinical characteristics of adolescents with rare genetic disorders with a behavior theory-driven approach.</p></div><div><h3>Method</h3><p>We investigated the behavioral and emotional problems and current and lifetime autistic traits in a sample of 93 Norwegian adolescents (<em>M</em><sub>age</sub> = 13.2 years, SD<sub>age</sub> = 2.4, range<sub>age</sub> 10–17, 62.4% females, 37.6% males) with various rare genetic disorders. The adolescents were investigated cross-sectionally utilizing standardized psychometric questionnaires rated by their parents.</p></div><div><h3>Results</h3><p>More current and lifetime autistic traits and social withdrawal were all associated with more internalizing problems. Further analyses demonstrated that social withdrawal partially mediated the positive association between current autistic traits and internalizing problems. In contrast, social withdrawal fully mediated the positive association between lifetime autistic traits and internalizing problems.</p></div><div><h3>Conclusion and implications</h3><p>Our results demonstrate important characteristics of adolescents with rare genetic disorders that may guide clinicians and future interventions. Social withdrawal may be prodromal to internalizing problems such as anxiety and depression. Thus, clinically addressing social withdrawal can represent a means to prevent internalizing problems in adolescents with rare genetic disorders and autistic traits.</p></div>\",\"PeriodicalId\":51351,\"journal\":{\"name\":\"Research in Developmental Disabilities\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":2.9000,\"publicationDate\":\"2024-02-16\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.sciencedirect.com/science/article/pii/S0891422224000313/pdfft?md5=37aa672a60cdce910355527aeffbc81a&pid=1-s2.0-S0891422224000313-main.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Research in Developmental Disabilities\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S0891422224000313\",\"RegionNum\":2,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q1\",\"JCRName\":\"EDUCATION, SPECIAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Research in Developmental Disabilities","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0891422224000313","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"EDUCATION, SPECIAL","Score":null,"Total":0}
Autism traits, social withdrawal, and behavioral and emotional problems in a Norwegian cohort of adolescents with rare genetic disorders
Background
Adolescents with rare genetic disorders represent a population that can be at risk in psychosocial terms. Despite its importance, the association of autism traits and social withdrawal in behavioral and emotional problems in adolescents with rare genetic disorders remains understudied.
Aim
The study aimed to empirically examine the clinical characteristics of adolescents with rare genetic disorders with a behavior theory-driven approach.
Method
We investigated the behavioral and emotional problems and current and lifetime autistic traits in a sample of 93 Norwegian adolescents (Mage = 13.2 years, SDage = 2.4, rangeage 10–17, 62.4% females, 37.6% males) with various rare genetic disorders. The adolescents were investigated cross-sectionally utilizing standardized psychometric questionnaires rated by their parents.
Results
More current and lifetime autistic traits and social withdrawal were all associated with more internalizing problems. Further analyses demonstrated that social withdrawal partially mediated the positive association between current autistic traits and internalizing problems. In contrast, social withdrawal fully mediated the positive association between lifetime autistic traits and internalizing problems.
Conclusion and implications
Our results demonstrate important characteristics of adolescents with rare genetic disorders that may guide clinicians and future interventions. Social withdrawal may be prodromal to internalizing problems such as anxiety and depression. Thus, clinically addressing social withdrawal can represent a means to prevent internalizing problems in adolescents with rare genetic disorders and autistic traits.
期刊介绍：
Research In Developmental Disabilities is aimed at publishing original research of an interdisciplinary nature that has a direct bearing on the remediation of problems associated with developmental disabilities. Manuscripts will be solicited throughout the world. Articles will be primarily empirical studies, although an occasional position paper or review will be accepted. The aim of the journal will be to publish articles on all aspects of research with the developmentally disabled, with any methodologically sound approach being acceptable.
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