Division of Brain Sciences, Department of Gene Therapy, Faculty of Medicine, Centre for Neuroinflammation & Neurodegeneration, Imperial College London, Hammersmith Hospital Campus, London, UK.
Mol Ther. 2013 Oct;21(10):1862-75. doi: 10.1038/mt.2013.132. Epub 2013 Jun 25.
Huntington's disease (HD) is a devastating neurodegenerative disorder caused by abnormal polyglutamine expansion in the huntingtin protein (Exp-Htt). Currently, there are no effective treatments for HD. We used bidirectional lentiviral transfer vectors to generate in vitro and in vivo models of HD and to test the therapeutic potential of vascular endothelial growth factor 165 (VEGF₁₆₅). Lentiviral-mediated expression of Exp-Htt caused cell death and aggregate formation in human neuroblastoma SH-SY5Y and rat primary striatal cultures. Lentiviral-mediated VEGF₁₆₅ expression was found to be neuroprotective in both of these models. Unilateral stereotaxic vector delivery of Exp-Htt vector in adult rat striatum led to progressive inclusion formation and striatal neuron loss at 10 weeks post-transduction. Coinjection of a lower dose VEGF₁₆₅ significantly attenuated DARPP-32(+) neuronal loss, enhanced NeuN staining and reduced Exp-Htt aggregation. A tenfold higher dose VEGF₁₆₅ led to overt neuronal toxicity marked by tissue damage, neovascularization, extensive astrogliosis, vascular leakage, chronic inflammation and distal neuronal loss. No overt behavioral phenotype was observed in these animals. Expression of VEGF₁₆₅ at this higher dose in the brain of wild-type rats led to early mortality with global neuronal loss. This report raises important safety concerns about unregulated VEGF₁₆₅ CNS applications.
亨廷顿病(HD)是一种由亨廷顿蛋白(Exp-Htt)中异常多聚谷氨酰胺扩展引起的毁灭性神经退行性疾病。目前,HD 尚无有效治疗方法。我们使用双向慢病毒转移载体,生成体外和体内 HD 模型,并测试血管内皮生长因子 165(VEGF₁₆₅)的治疗潜力。慢病毒介导的 Exp-Htt 表达导致人神经母细胞瘤 SH-SY5Y 和大鼠原代纹状体培养物中的细胞死亡和聚集形成。在这两种模型中,慢病毒介导的 VEGF₁₆₅表达均具有神经保护作用。在成年大鼠纹状体中进行单侧立体定位载体传递 Exp-Htt 载体,导致转导后 10 周时包涵体形成和纹状体神经元丢失的进行性进展。共注射较低剂量的 VEGF₁₆₅可显著减轻 DARPP-32(+)神经元丢失,增强 NeuN 染色并减少 Exp-Htt 聚集。十倍高剂量的 VEGF₁₆₅导致明显的神经元毒性,表现为组织损伤、新生血管形成、广泛的星形胶质细胞增生、血管渗漏、慢性炎症和远端神经元丢失。这些动物没有观察到明显的行为表型。在野生型大鼠大脑中以更高剂量表达 VEGF₁₆₅会导致早期死亡和全脑神经元丢失。该报告提出了关于不受监管的 VEGF₁₆₅ 中枢神经系统应用的重要安全问题。