Roy Choudhury Nilpawan, Hilber Pascal, Cendelin Jan
Department of Pathological Physiology, Faculty of Medicine in Pilsen, Charles University, Pilsen, Czech Republic.
Univ Rouen Normandie, Inserm, Normandie Univ, CBG UMR 1245 NeuroGlio Team, Rouen, France.
Cerebellum. 2025 Feb 28;24(2):54. doi: 10.1007/s12311-025-01810-5.
Cerebellar extinction lesions can manifest themselves with cerebellar motor and cerebellar cognitive affective syndromes. For investigation of the functions of the cerebellum and the pathogenesis of cerebellar diseases, particularly hereditary neurodegenerative cerebellar ataxias, various cerebellar mutant mice are used. The Lurcher mouse is a model of selective olivocerebellar degeneration with early onset and rapid progress. These mice show both motor deficits as well as cognitive and behavioral changes i.e., pathological phenotype in the functional domains affected in cerebellar patients. Therefore, Lurcher mice might be considered as a tool to investigate the mechanisms of functional impairments caused by cerebellar degenerative diseases. There are, however, limitations due to the particular features of the neurodegenerative process and a lack of possibilities to examine some processes in mice. The main advantage of Lurcher mice would be the expected absence of significant neuropathologies outside the olivocerebellar system that modify the complex behavioral phenotype in less selective models. However, detailed examinations and further thorough validation of the model are needed to verify this assumption.
小脑消退性病变可表现为小脑运动综合征和小脑认知情感综合征。为了研究小脑的功能以及小脑疾病,特别是遗传性神经退行性小脑共济失调的发病机制,人们使用了各种小脑突变小鼠。Lurcher小鼠是一种选择性橄榄小脑变性的模型,具有发病早、进展快的特点。这些小鼠既表现出运动缺陷,也表现出认知和行为变化,即小脑疾病患者受影响功能领域的病理表型。因此,Lurcher小鼠可被视为研究小脑退行性疾病所致功能障碍机制的一种工具。然而,由于神经退行性过程的特殊特征以及在小鼠中缺乏检查某些过程的可能性,存在一定局限性。Lurcher小鼠的主要优势在于,预计在橄榄小脑系统之外不会出现显著的神经病理学改变,而在选择性较低的模型中,这些改变会改变复杂的行为表型。然而,需要进行详细检查和对该模型进行进一步深入验证,以证实这一假设。