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前额叶皮质中钙视网膜蛋白的缺乏会导致小鼠出现与自闭症谱系障碍相关的行为缺陷。

Deficiency of calretinin in prefrontal cortex causes behavioral deficits relevant to autism spectrum disorder in mice.

作者信息

Zhang Yaodong, Zhao Xiaotong, Gao Chao, Shi Shengli, Chen Mengyuan, Guo Bin, Hu Shunan, Mei Daoqi, Duan Xujun, Wang Xiaona

机构信息

Henan Children's Hospital, Henan Key Laboratory of Children's Genetics and Metabolic Diseases, Henan Children's Neurodevelopment Engineering Research Center, Children's Hospital Affiliated to Zhengzhou University, Zhengzhou Children's Hospital, Zhengzhou, China.

Department of Rehabilitation, Children's Hospital Affiliated to Zhengzhou University, Zhengzhou, China.

出版信息

Mol Brain. 2025 Jul 12;18(1):61. doi: 10.1186/s13041-025-01233-7.

Abstract

Autism spectrum disorder (ASD) is a neurodevelopmental disorder characterized by core symptoms including deficits in social interaction, repetitive and stereotyped behaviors, along with higher levels of anxiety and cognitive impairments. Previous studies demonstrate pronounced reduced density of calretinin (CR)-expressing GABAergic interneurons in both ASD patients and animal models. The object of the current study was to determine the role of CR in ASD-relevant behavioral aberrations. Herein, the mRNA and protein levels of CR in the prefrontal cortex (PFC) of mouse model of ASD based on prenatal exposure to valproic acid (VPA) were determined by qRT-PCR and Western blot analysis, respectively. Moreover, the behavioral abnormalities in naive mice with CR deficiency mediated by recombinant adeno-associated virus (rAAV) were evaluated in a comprehensive testing battery including social interaction, marble burying, self-grooming, open-field, elevated plus maze and novel object recognition tests. Furthermore, the action potential changes caused by CR deficiency were examined in neurons within the PFC in naive mouse. The results show that the mRNA and protein levels of PFC CR of VPA-induced mouse ASD model were reduced. Concomitantly, mice with CR knockdown displayed ASD-like behavioral aberrations, such as social impairments, elevated stereotypes, anxiety and memory defects. Intriguingly, patch-clamp recordings revealed that CR knockdown provoked decreased neuronal excitability by increasing action potential discharge frequencies together with decreased action potential threshold and rheobase. Our findings support a notion that CR knockdown might contribute to ASD-like phenotypes, with the pathogenesis most likely stemming from increased neuronal excitability.

摘要

自闭症谱系障碍(ASD)是一种神经发育障碍,其核心症状包括社交互动缺陷、重复刻板行为,以及较高水平的焦虑和认知障碍。先前的研究表明,ASD患者和动物模型中表达钙视网膜蛋白(CR)的γ-氨基丁酸能中间神经元密度均显著降低。本研究的目的是确定CR在与ASD相关的行为异常中的作用。在此,分别通过qRT-PCR和蛋白质印迹分析,测定了基于产前暴露于丙戊酸(VPA)的ASD小鼠模型前额叶皮质(PFC)中CR的mRNA和蛋白质水平。此外,利用重组腺相关病毒(rAAV)介导的CR缺陷的新生小鼠,在包括社交互动、大理石掩埋、自我梳理、旷场、高架十字迷宫和新物体识别测试在内的综合测试中,评估了其行为异常。此外,还检测了新生小鼠PFC内神经元因CR缺陷引起的动作电位变化。结果显示,VPA诱导的小鼠ASD模型PFC中CR的mRNA和蛋白质水平降低。同时,CR基因敲低的小鼠表现出类似ASD的行为异常,如社交障碍、刻板行为增加、焦虑和记忆缺陷。有趣的是,膜片钳记录显示,CR基因敲低通过增加动作电位发放频率以及降低动作电位阈值和阈强度,导致神经元兴奋性降低。我们的研究结果支持这样一种观点,即CR基因敲低可能导致类似ASD的表型,其发病机制很可能源于神经元兴奋性增加。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c862/12255998/a8ed2c18c02f/13041_2025_1233_Fig1_HTML.jpg

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