{"title":"Compensatory synaptotagmin-11 expression conceals parkinson's disease-like phenotypes in parkin knockout mice.","authors":"Nan Dong, Zhenli Xie, Anqi Wei, Yuxin Yang, Yongning Deng, Xu Cheng, Bianbian Wang, Yang Chen, Yuhao Gu, Jingyu Yao, Yuhao Qin, Chaowen Zheng, Xi Zhang, Yuqing Zhang, Xinjiang Kang, Guoqing Chen, Qiumin Qu, Changhe Wang, Huadong Xu","doi":"10.1186/s12964-025-02037-x","DOIUrl":null,"url":null,"abstract":"<p><p>Animal models are crucial for elucidating the pathological mechanisms underlying Parkinson's disease (PD). Unfortunately, most of transgenic mouse models fail to manifest pathological changes observed in PD patients, pending the advancement of PD research. However, the mechanism underlying this discrepancy remains elusive. Here, we provide compelling evidence that the compensatory expression of synaptotagmin-11 (Syt11) plays a key role in concealing PD-associated phenotypes in parkin knockout (KO) mouse models. Unlike the normal dopamine (DA) release and motor behaviors observed in parkin KO mice, parkin knockdown (KD) in the substantia nigra pars compacta (SNpc) in adult mice led to both the impaired DA release and the pronounced motor deficits. Interestingly, Syt11, a well-established parkin substrate involved in PD, was specifically upregulated in parkin KD mice and in parkin KO mice during the suckling stage, but not in adult parkin KO mice. Importantly, the overexpression of Syt11 alone is capable of inducing PD-like motor and non-motor impairments, as well as the impaired DA release and reuptake, which is essential for parkin-associated pathogenesis of PD. Therefore, this work not only elucidate a compensatory mechanism that accounts for the absence of overt PD phenotypes in parkin KO mice, but also contribute to the comprehensive understanding of the progression of PD, opening new avenues for the therapeutic treatment of PD.</p>","PeriodicalId":55268,"journal":{"name":"Cell Communication and Signaling","volume":"23 1","pages":"61"},"PeriodicalIF":8.2000,"publicationDate":"2025-02-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11789404/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Cell Communication and Signaling","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1186/s12964-025-02037-x","RegionNum":2,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"CELL BIOLOGY","Score":null,"Total":0}
引用次数: 0
摘要
动物模型对于阐明帕金森病(PD)的病理机制至关重要。遗憾的是,大多数转基因小鼠模型都无法表现出帕金森病患者的病理变化,这阻碍了帕金森病研究的进展。然而,造成这种差异的机制仍然难以捉摸。在这里,我们提供了令人信服的证据,证明突触标记蛋白-11(Syt11)的代偿性表达在掩盖帕金敲除(KO)小鼠模型中与帕金症相关的表型方面发挥了关键作用。与在 Parkin KO 小鼠中观察到的正常多巴胺(DA)释放和运动行为不同,在成年小鼠的黑质紧凑部(SNpc)中敲除 Parkin 会导致 DA 释放受损和明显的运动障碍。有趣的是,在parkin KD小鼠和parkin KO小鼠的哺乳期,Syt11(一种已被证实与帕金森病有关的parkin底物)特异性上调,而在成年parkin KO小鼠中则没有上调。重要的是,单独过表达 Syt11 能够诱导类似于帕金森病的运动和非运动障碍,以及 DA 释放和再摄取受损,而 DA 释放和再摄取受损对于帕金相关的帕金森病发病机制至关重要。因此,这项工作不仅阐明了parkin KO小鼠没有明显的帕金森病表型的代偿机制,而且有助于全面了解帕金森病的进展,为帕金森病的治疗开辟了新途径。
Animal models are crucial for elucidating the pathological mechanisms underlying Parkinson's disease (PD). Unfortunately, most of transgenic mouse models fail to manifest pathological changes observed in PD patients, pending the advancement of PD research. However, the mechanism underlying this discrepancy remains elusive. Here, we provide compelling evidence that the compensatory expression of synaptotagmin-11 (Syt11) plays a key role in concealing PD-associated phenotypes in parkin knockout (KO) mouse models. Unlike the normal dopamine (DA) release and motor behaviors observed in parkin KO mice, parkin knockdown (KD) in the substantia nigra pars compacta (SNpc) in adult mice led to both the impaired DA release and the pronounced motor deficits. Interestingly, Syt11, a well-established parkin substrate involved in PD, was specifically upregulated in parkin KD mice and in parkin KO mice during the suckling stage, but not in adult parkin KO mice. Importantly, the overexpression of Syt11 alone is capable of inducing PD-like motor and non-motor impairments, as well as the impaired DA release and reuptake, which is essential for parkin-associated pathogenesis of PD. Therefore, this work not only elucidate a compensatory mechanism that accounts for the absence of overt PD phenotypes in parkin KO mice, but also contribute to the comprehensive understanding of the progression of PD, opening new avenues for the therapeutic treatment of PD.
期刊介绍:
Cell Communication and Signaling (CCS) is a peer-reviewed, open-access scientific journal that focuses on cellular signaling pathways in both normal and pathological conditions. It publishes original research, reviews, and commentaries, welcoming studies that utilize molecular, morphological, biochemical, structural, and cell biology approaches. CCS also encourages interdisciplinary work and innovative models, including in silico, in vitro, and in vivo approaches, to facilitate investigations of cell signaling pathways, networks, and behavior.
Starting from January 2019, CCS is proud to announce its affiliation with the International Cell Death Society. The journal now encourages submissions covering all aspects of cell death, including apoptotic and non-apoptotic mechanisms, cell death in model systems, autophagy, clearance of dying cells, and the immunological and pathological consequences of dying cells in the tissue microenvironment.