贝克维茨-韦德曼综合症儿童和青少年的认知、社交和情感行为结果。

IF 1.6 3区 医学 Q3 GENETICS & HEREDITY
Niccolò Butti, Cosimo Urgesi, Alessandro Mussa, Rosario Montirosso
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引用次数: 0

摘要

虽然贝克维茨-韦德曼综合征谱系(BWSp)通常与智力障碍无关,但最近的证据表明,需要进一步研究贝克维茨-韦德曼综合征谱系儿童的认知发展和学习技能。此外,研究还记录了与 BWSp 相关的社交困难和情绪行为问题。然而,目前仍缺乏对 BWSp 儿童社会情感发展的全面描述。在本研究中,我们使用非语言智力测验、涵盖多个领域的神经心理测试、学习技能测试以及评估自闭症特征和情绪行为问题的问卷,对 29 名 5-18 岁的 BWSp 儿童的认知和社会情感发展进行了评估。不出所料,大多数参与者在认知测试中都表现出了足够的能力。然而,研究结果也突出表明,语言方面的困难大于视觉空间处理能力,社会感知能力较强,阅读和心算能力较慢。对情绪行为障碍的评估表明,自闭症患者普遍存在焦虑增加、自卑、社交退缩和倾向于控制外化反应等表型,但与自闭症特征、认知结果和近期共识声明提出的临床评分没有关联。社交感知和内化问题的增加可能是应对社交和护理相关压力的策略所致。总之,本研究的结果可为 BWSp 儿童和青少年的临床管理和遗传咨询提供参考。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Cognitive, Social, and Emotional-Behavioral Outcomes in Children and Adolescents With Beckwith-Wiedemann Syndrome.

Although Beckwith-Wiedemann syndrome spectrum (BWSp) is not usually associated with intellectual disability, recent evidences calls for further investigation of cognitive development and academic skills in children with BWSp. Moreover, research has documented social difficulties and emotional-behavioral problems associated with BWSp. Nevertheless, a full characterization of socio-emotional development in BWSp is still lacking. In the current study, cognitive and socio-emotional development was assessed in 29 children with BWSp aged 5-18 years, using a test of nonverbal intelligence, a neuropsychological battery covering multiple domains, academic skills tests, and questionnaires evaluating autistic traits and emotional-behavioral problems. As expected, most participants showed adequate performance in cognitive tests. However, the findings also highlighted greater difficulties in language than visuospatial processing, strengths in social perception, as well as slowness in reading and mental calculation. The assessment of emotional-behavioral difficulties indicated a prevalent phenotype characterized by increased anxiety, low self-esteem, social withdrawal and a tendency to control externalizing reactions, but no associations with autistic traits, cognitive outcomes, and the clinical score proposed by the recent Consensus statement. Increased social perception and internalization problems likely result from coping strategies with social and care-related stress. Overall, the findings of this study inform clinical management and genetic counseling for children and adolescents with BWSp.

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来源期刊
CiteScore
5.90
自引率
7.10%
发文量
40
审稿时长
4-8 weeks
期刊介绍: Neuropsychiatric Genetics, Part B of the American Journal of Medical Genetics (AJMG) , provides a forum for experimental and clinical investigations of the genetic mechanisms underlying neurologic and psychiatric disorders. It is a resource for novel genetics studies of the heritable nature of psychiatric and other nervous system disorders, characterized at the molecular, cellular or behavior levels. Neuropsychiatric Genetics publishes eight times per year.
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