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自闭症样Shank3B基因缺陷小鼠中初级多巴胺能神经元的神经突分支减少

Reduced Neurite Arborization in Primary Dopaminergic Neurons in Autism-Like Shank3B-Deficient Mice.

作者信息

Bacova Zuzana, Havranek Tomas, Mihalj Denisa, Borbelyova Veronika, Kostrubanicova Kristina, Kramarova Michaela, Ostatnikova Daniela, Bakos Jan

机构信息

Institute of Experimental Endocrinology, Biomedical Research Center, Slovak Academy of Sciences, Bratislava, Slovakia.

Institute of Anatomy, Faculty of Medicine, Comenius University in Bratislava, Bratislava, Slovakia.

出版信息

Mol Neurobiol. 2025 May;62(5):5838-5849. doi: 10.1007/s12035-024-04652-0. Epub 2024 Dec 9.

DOI:10.1007/s12035-024-04652-0
PMID:39654001
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11953188/
Abstract

Despite many studies on dopamine changes in autism, specific alterations in midbrain dopamine neurons projecting to the striatum and cortex remain unclear. Mouse models with diverse SH3 domain and ankyrin repeat containing protein 3 (Shank3) deficiencies are used for investigating autistic symptoms and underlying neurobiological mechanisms. SHANK3 belongs to postsynaptic proteins crucial for synapse formation during development, and disruptions in SHANK3 structure could lead to impaired neurite outgrowth and altered dendritic arborization and morphology. Therefore, we aimed to investigate whether Shank3 deficiency (Shank3B) leads to changes in the morphology of primary neuronal cell cultures from dopaminergic brain regions of neonatal mouse pups and whether it results in alterations in synaptic proteins in dopaminergic nerve pathway projection areas (striatum, frontal cortex). Significantly reduced neurite outgrowth was observed in primary dopaminergic neurons from the midbrain and striatum of Shank3-deficient compared to WT mice. A decrease in Synapsin I immunofluorescence signal in the cortical neurons isolated from Shank3-deficient mice was found, although neurite arborization changes were less severe. Importantly, the deficit in the length of the longest neurite was confirmed in primary cortical neurons isolated from Shank3-deficient mice. No changes in the gene expression of synaptic proteins were observed in the striatum and frontal cortex of Shank3-deficient mice, but an altered gene expression profile of dopaminergic receptors was found. These results show structural changes of dopaminergic neurons, which may explain autistic symptomatology in the used model and provide a basis for understanding the long-term development of autistic symptoms.

摘要

尽管针对自闭症中多巴胺变化进行了许多研究,但投射到纹状体和皮层的中脑多巴胺神经元的具体改变仍不清楚。具有多种含SH3结构域和锚蛋白重复序列蛋白3(Shank3)缺陷的小鼠模型被用于研究自闭症症状及潜在的神经生物学机制。SHANK3属于发育过程中对突触形成至关重要的突触后蛋白,SHANK3结构的破坏可能导致神经突生长受损以及树突分支和形态改变。因此,我们旨在研究Shank3缺陷(Shank3B)是否会导致新生小鼠幼崽多巴胺能脑区原代神经元细胞培养物的形态变化,以及是否会导致多巴胺能神经通路投射区域(纹状体、额叶皮层)中突触蛋白的改变。与野生型小鼠相比,在Shank3缺陷小鼠中脑和纹状体的原代多巴胺能神经元中观察到神经突生长显著减少。虽然树突分支变化不太严重,但在从Shank3缺陷小鼠分离的皮层神经元中发现突触素I免疫荧光信号减少。重要的是,在从Shank3缺陷小鼠分离的原代皮层神经元中证实了最长神经突长度的缺陷。在Shank3缺陷小鼠的纹状体和额叶皮层中未观察到突触蛋白基因表达的变化,但发现多巴胺能受体的基因表达谱发生了改变。这些结果显示了多巴胺能神经元的结构变化,这可能解释了所使用模型中的自闭症症状,并为理解自闭症症状的长期发展提供了基础。

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Am J Pathol. 2024 Jun;194(6):1062-1077. doi: 10.1016/j.ajpath.2024.02.014. Epub 2024 Mar 16.
2
Neuron type-specific proteomics reveals distinct Shank3 proteoforms in iSPNs and dSPNs lead to striatal synaptopathy in Shank3B mice.神经元类型特异性蛋白质组学揭示 iSPNs 和 dSPNs 中独特的 Shank3 蛋白形式,导致 Shank3B 小鼠纹状体突触病变。
Mol Psychiatry. 2024 Aug;29(8):2372-2388. doi: 10.1038/s41380-024-02493-w. Epub 2024 Mar 14.
3
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自闭症谱系障碍的多巴胺假说:证据的综合分析。
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