Department of Child and Adolescent Psychiatry, Psychotherapy and Psychosomatics, University Medical Center Hamburg, Eppendorf, Germany.
Division Mental Health Care, Innlandet Hospital Trust, Norway; Department of Psychology, Inland Norway University of Applied Sciences, Norway.
Res Dev Disabil. 2024 Apr;147:104699. doi: 10.1016/j.ridd.2024.104699. Epub 2024 Feb 16.
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.
The study aimed to empirically examine the clinical characteristics of adolescents with rare genetic disorders with a behavior theory-driven approach.
We investigated the behavioral and emotional problems and current and lifetime autistic traits in a sample of 93 Norwegian adolescents (M = 13.2 years, SD = 2.4, range 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.
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.
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.
患有罕见遗传疾病的青少年在心理社会方面存在风险。尽管这一点很重要,但自闭症特征与罕见遗传疾病青少年的行为和情绪问题中的社交退缩之间的关联仍研究不足。
本研究旨在采用行为理论驱动的方法,对患有罕见遗传疾病的青少年的临床特征进行实证研究。
我们调查了 93 名挪威青少年(M = 13.2 岁,SD = 2.4,范围 10-17,62.4%为女性,37.6%为男性)的行为和情绪问题以及当前和终身自闭症特征,他们患有各种罕见遗传疾病。利用父母评定的标准化心理计量问卷对青少年进行了横断面调查。
更多的当前和终身自闭症特征和社交退缩均与更多的内化问题相关。进一步的分析表明,社交退缩部分中介了当前自闭症特征与内化问题之间的正相关。相比之下,社交退缩完全中介了终身自闭症特征与内化问题之间的正相关。
我们的研究结果展示了罕见遗传疾病青少年的重要特征,这些特征可能为临床医生和未来的干预措施提供指导。社交退缩可能是焦虑和抑郁等内化问题的先兆。因此,临床上解决社交退缩问题可能是预防罕见遗传疾病和自闭症特征青少年内化问题的一种手段。