Ras 通路疾病中的社会交流:神经纤维瘤病 1 型和努南综合征从遗传到行为的全面回顾。

IF 9.6 1区 医学 Q1 NEUROSCIENCES
Monica Siqueiros-Sanchez, Yaffa Serur, Chloe A McGhee, Taylor F Smith, Tamar Green
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引用次数: 0

摘要

神经纤维瘤病 1 型(NF1)和努南综合征(NS)是由编码 Ras-ERK-MAPK 信号通路(Ras 通路)成分的致病变体引起的神经遗传综合征。NF1 和 NS 与社会交流差异和相关的神经精神风险有关。在过去十年中,人们越来越关注将 Ras 相关综合征作为了解社会交流障碍和自闭症谱系障碍的模型。我们系统地回顾了 2010-2023 年间的文献,重点关注 RDoC 框架中的社会交流构建。我们对 NF1 和 NS 中面部和非面部社会交流过程的研究进行了综合总结,涉及分子、细胞、神经回路和行为等多个领域。在分子和细胞水平上,Ras通路的失调通过GABA能、谷氨酸能和5-羟色胺能传导的变化以及抑制/兴奋失衡与社交沟通的变化密切相关。在 NF1 和 NS 中,通常与学习、注意力和记忆有关的神经回路(如皮质纹状体连接)也与社交沟通有关。我们强调了研究较少的潜在社会交流机制,如白质连接和默认模式网络。最后,我们指出了 NF1 和 NS 文献中的主要空白,并提供了未来研究的路线图。通过利用遗传综合征研究,我们可以了解与行为和精神障碍相关的机制。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Social Communication in Ras Pathway Disorders: A Comprehensive Review From Genetics to Behavior in Neurofibromatosis Type 1 and Noonan Syndrome.

Neurofibromatosis type 1 (NF1) and Noonan syndrome (NS) are neurogenetic syndromes caused by pathogenetic variants encoding components of the Ras-ERK-MAPK (Ras/extracellular signal-regulated kinase/mitogen-activated protein kinase) signaling pathway (Ras pathway). NF1 and NS are associated with differences in social communication and related neuropsychiatric risks. During the last decade, there has been growing interest in Ras-linked syndromes as models to understand social communication deficits and autism spectrum disorder. We systematically review the literature between 2010 and 2023 focusing on the social communication construct of the Research Domain Criteria framework. We provide an integrative summary of the research on facial and nonfacial social communication processes in NF1 and NS across molecular, cellular, neural circuitry, and behavioral domains. At the molecular and cellular levels, dysregulation in the Ras pathway is intricately tied to variations in social communication through changes in GABAergic (gamma-aminobutyric acidergic), glutamatergic, and serotonergic transmission, as well as inhibitory/excitatory imbalance. Neural circuitry typically associated with learning, attention, and memory in NF1 and NS (e.g., corticostriatal connectivity) is also implicated in social communication. We highlight less-researched potential mechanisms for social communication, such as white matter connectivity and the default mode network. Finally, key gaps in NF1 and NS literature are identified, and a roadmap for future research is provided. By leveraging genetic syndrome research, we can understand the mechanisms associated with behaviors and psychiatric disorders.

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来源期刊
Biological Psychiatry
Biological Psychiatry 医学-精神病学
CiteScore
18.80
自引率
2.80%
发文量
1398
审稿时长
33 days
期刊介绍: Biological Psychiatry is an official journal of the Society of Biological Psychiatry and was established in 1969. It is the first journal in the Biological Psychiatry family, which also includes Biological Psychiatry: Cognitive Neuroscience and Neuroimaging and Biological Psychiatry: Global Open Science. The Society's main goal is to promote excellence in scientific research and education in the fields related to the nature, causes, mechanisms, and treatments of disorders pertaining to thought, emotion, and behavior. To fulfill this mission, Biological Psychiatry publishes peer-reviewed, rapid-publication articles that present new findings from original basic, translational, and clinical mechanistic research, ultimately advancing our understanding of psychiatric disorders and their treatment. The journal also encourages the submission of reviews and commentaries on current research and topics of interest.
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