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维甲酸治疗自闭症改善社会行为的机制洞察:来自大鼠多组学研究的证据

Mechanistic insights into retinoic-acid treatment for autism in the improvement of social behavior: Evidence from a multi omics study in rats.

作者信息

Zhu Jiang, Liu Huan, Hu Yan, Liu Juan, Dai Chunfang, Liang Jingjing, Cheng Boli, Tan Mei, Zhang Yaoyin, Cao Qingjiu, Lai Xi

机构信息

Department of Child Health Care, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangdong Provincial Clinical Research Center for Child Health, Guangzhou, 510623, China.

Mianyang Key Laboratory of Anesthesia and Neuroregulation, Department of Anesthesiology, Mianyang Central Hospital, Mianyang, 621000, China; Department of Pediatrics, Mianyang Central Hospital, School of Medicine, University of Electronic Science and Technology of China, Mianyang, 621000, China.

出版信息

Neuropharmacology. 2025 Mar 1;265:110244. doi: 10.1016/j.neuropharm.2024.110244. Epub 2024 Dec 4.

Abstract

BACKGROUND

Autism spectrum disorder (ASD) is a lifelong condition. It is characterized by complex etiologies, including disruptions in exogenous retinoic acid (RA) signaling, which may serve as an environmental risk factor. Targeting the RA pathway presents a promising therapeutic avenue, though the precise mechanisms remain to be elucidated.

METHODS

Female Sprague-Dawley rats were treated with valproic acid (VPA) during pregnancy to induce an ASD model in their offspring. Some offspring received RA treatment postnatally. Social behavior and brain-functional connectivity were assessed using behavioral tests and functional magnetic resonance imaging (fMRI), respectively. Transcriptomics analysis and proteomics analysis of the hypothalamus identified differentially expressed genes (DEGs) and differentially expressed proteins (DEPs). These were intersected with ASD pathogenic genes (APGs) and ASD pathogenic proteins (APPs) to identify differentially expressed APGs (DE-APGs) and differentially expressed APPs (DE-APPs), which were validated by real-time reverse transcription-quantitative polymerase chain reaction (RT-qPCR) and western blotting. Analyses of enrichment of signaling pathways were done using the Kyoto Encyclopedia of Genes and Genomes database.

RESULTS

RA treatment significantly improved social behaviors and revealed distinct patterns of hypo- and hyper-connectivity across various brain regions, with notable changes involving the hypothalamus and facial nerve. Differential analysis revealed 4165 DEGs (DEG 1) and 329 DEPs (DEP 1) between control and VPA groups, and 1610 DEGs (DEG 2) and 197 DEPs (DEP 2) between VPA and RA supplementation (RAS) groups. Twenty-two DE-APGs and five DE-APPs were identified, with key associations found between proteins such as Tbl1xr1 and Myo5a and >13 genes including Nrxn1, Cacna1e, and Gabrb2. Significant alterations in DE-APGs, including Grin2b, Nrxn1, Cacna1e, and Gabrb2, were confirmed via real-time RT-PCR and western blotting. In addition, 22 key signaling pathways were enriched in DEPs and DEGs.

CONCLUSION

RA supplementation in ASD rats induced by VPA may ameliorate social deficits and modulated functional connectivity, especially in the hypothalamus and facial nerve regions. This suggests potential therapeutic benefits for neural circuitry dysregulation in ASD. Additionally, RA altered critical gene and protein expressions in hypothalamus, implicating its role in modulating key signaling pathways to mitigate social deficits in ASD. This study provides new insights into the molecular mechanisms of ASD and supports the development of novel therapeutic strategies.

摘要

背景

自闭症谱系障碍(ASD)是一种终身疾病。其病因复杂,包括外源性视黄酸(RA)信号传导中断,这可能是一种环境风险因素。尽管确切机制仍有待阐明,但针对RA途径是一条有前景的治疗途径。

方法

在孕期用丙戊酸(VPA)处理雌性斯普拉格-道利大鼠,以在其后代中诱导出ASD模型。一些后代在出生后接受RA治疗。分别使用行为测试和功能磁共振成像(fMRI)评估社交行为和脑功能连接性。对下丘脑进行转录组学分析和蛋白质组学分析,以鉴定差异表达基因(DEGs)和差异表达蛋白质(DEPs)。将这些与ASD致病基因(APGs)和ASD致病蛋白质(APPs)进行交叉分析,以鉴定差异表达的APGs(DE-APGs)和差异表达的APPs(DE-APPs),并通过实时逆转录定量聚合酶链反应(RT-qPCR)和蛋白质免疫印迹法进行验证。使用京都基因与基因组百科全书数据库对信号通路的富集情况进行分析。

结果

RA治疗显著改善了社交行为,并揭示了不同脑区低连接性和高连接性的不同模式,其中下丘脑和面神经有明显变化。差异分析显示,对照组和VPA组之间有4165个DEGs(DEG 1)和329个DEPs(DEP 1),VPA组和补充RA(RAS)组之间有1610个DEGs(DEG 2)和197个DEPs(DEP 2)。鉴定出22个DE-APGs和5个DE-APPs,发现Tbl1xr1和Myo5a等蛋白质与包括Nrxn1、Cacna1e和Gabrb2在内的13个以上基因之间存在关键关联。通过实时RT-PCR和蛋白质免疫印迹法证实了DE-APGs的显著变化,包括Grin2b、Nrxn1、Cacna1e和Gabrb2。此外,22条关键信号通路在DEPs和DEGs中富集。

结论

VPA诱导的ASD大鼠补充RA可能改善社交缺陷并调节功能连接性,尤其是在下丘脑和面神经区域。这表明对ASD中神经回路失调具有潜在的治疗益处。此外,RA改变了下丘脑中关键基因和蛋白质的表达,暗示其在调节关键信号通路以减轻ASD社交缺陷中的作用。本研究为ASD的分子机制提供了新见解,并支持新型治疗策略的开发。

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