Pérez-Revuelta Laura, Pérez-Boyero David, Pérez-Martín Ester, Cabedo Valeria Lorena, Téllez de Meneses Pablo González, Weruaga Eduardo, Díaz David, Alonso José Ramón
Laboratory of Neuronal Plasticity and Neurorepair, Institute of Neuroscience of Castile and Leon (INCyL), Universidad de Salamanca, 37007 Salamanca, Spain.
Institute of Biomedical Research of Salamanca (IBSAL), 37007 Salamanca, Spain.
Int J Mol Sci. 2025 Jan 10;26(2):538. doi: 10.3390/ijms26020538.
In recent decades, the scientific community has faced a major challenge in the search for new therapies that can slow down or alleviate the process of neuronal death that accompanies neurodegenerative diseases. This study aimed to identify an effective therapy using neurotrophic factors to delay the rapid and aggressive cerebellar degeneration experienced by the Purkinje Cell Degeneration (PCD) mouse, a model of childhood-onset neurodegeneration with cerebellar atrophy (CONDCA). Initially, we analyzed the changes in the expression of several neurotrophic factors related to the degenerative process itself, identifying changes in insulin-like growth factor 1 (IGF-1) and Vascular Endothelial Growth Factor B (VEGF-B) in the affected animals. Then, we administered pharmacological treatments using human recombinant IGF-1 (rhIGF-1) or VEGF-B (rhVEGF-B) proteins, considering their temporal variations during the degenerative process. The effects of these treatments on motor, cognitive, and social behavior, as well as on cerebellar destructuration were analyzed. Whereas treatment with rhIGF-1 did not demonstrate any neuroprotective effect, rhVEGF-B administration at moderate dosages stopped the process of neuronal death and restored motor, cognitive, and social functions altered in PCD mice (and CONDCA patients). However, increasing the frequency of rhVEGF-B administration had a detrimental effect on Purkinje cell survival, suggesting an inverted U-shaped dose-response curve of this substance. Additionally, we demonstrate that this neuroprotective effect was achieved through a partial inhibition or delay of apoptosis. These findings provide strong evidence supporting the use of rhVEGF-B as a pharmacological agent to limit severe cerebellar neurodegenerative processes.
近几十年来,科学界在寻找能够减缓或缓解伴随神经退行性疾病的神经元死亡过程的新疗法方面面临着重大挑战。本研究旨在确定一种使用神经营养因子的有效疗法,以延缓浦肯野细胞变性(PCD)小鼠所经历的快速且侵袭性的小脑变性,PCD小鼠是一种伴有小脑萎缩的儿童期神经退行性疾病(CONDCA)模型。最初,我们分析了与变性过程本身相关的几种神经营养因子表达的变化,确定了受影响动物中胰岛素样生长因子1(IGF-1)和血管内皮生长因子B(VEGF-B)的变化。然后,我们使用人重组IGF-1(rhIGF-1)或VEGF-B(rhVEGF-B)蛋白进行药物治疗,并考虑它们在变性过程中的时间变化。分析了这些治疗对运动、认知和社交行为以及小脑结构破坏的影响。虽然rhIGF-1治疗未显示出任何神经保护作用,但中等剂量的rhVEGF-B给药阻止了神经元死亡过程,并恢复了PCD小鼠(以及CONDCA患者)中改变的运动、认知和社交功能。然而,增加rhVEGF-B给药频率对浦肯野细胞存活有不利影响,表明该物质存在倒U形剂量反应曲线。此外,我们证明这种神经保护作用是通过部分抑制或延迟细胞凋亡实现的。这些发现提供了有力证据,支持将rhVEGF-B用作限制严重小脑神经退行性过程的药物制剂。