PTC Therapeutics, Mountain View, California, United States of America.
PTC Therapeutics, Warren, New Jersey, United States of America.
PLoS One. 2024 Sep 18;19(9):e0309893. doi: 10.1371/journal.pone.0309893. eCollection 2024.
Parkinson's disease is the second most common neurodegenerative disorder, affecting nearly 10 million people worldwide. Ferroptosis, a recently identified form of regulated cell death characterized by 15-lipoxygenase-mediated hydroperoxidation of membrane lipids, has been implicated in neurodegenerative disorders including amyotrophic lateral sclerosis and Parkinson's disease. Pharmacological inhibition of 15 -lipoxygenase to prevent iron- and lipid peroxidation-associated ferroptotic cell death is a rational strategy for the treatment of Parkinson's disease. We report here the characterization of PTC-041 as an anti-ferroptotic reductive lipoxygenase inhibitor developed for the treatment of Parkinson's disease. In these studies, PTC-041 potently protects primary human Parkinson's disease patient-derived fibroblasts from lipid peroxidation and subsequent ferroptotic cell death and prevents ferroptosis-related neuronal loss and astrogliosis in primary rat neuronal cultures. Additionally, PTC-041 prevents ferroptotic-mediated α-synuclein protein aggregation and nitrosylation in vitro, suggesting a potential role for anti-ferroptotic lipoxygenase inhibitors in mitigating pathogenic aspects of synucleinopathies such as Parkinson's disease. We further found that PTC-041 protects against synucleinopathy in vivo, demonstrating that PTC-041 treatment of Line 61 transgenic mice protects against α-synuclein aggregation and phosphorylation as well as prevents associated neuronal and non-neuronal cell death. Finally, we show that. PTC-041 protects against 6-hydroxydopamine-induced motor deficits in a hemiparkinsonian rat model, further validating the potential therapeutic benefits of lipoxygenase inhibitors in the treatment of Parkinson's disease.
帕金森病是第二常见的神经退行性疾病,影响全球近 1000 万人。铁死亡是一种新发现的受调控的细胞死亡形式,其特征是 15-脂氧合酶介导的膜脂质过氧化物化,已被认为与包括肌萎缩侧索硬化症和帕金森病在内的神经退行性疾病有关。抑制 15-脂氧合酶以防止铁和脂质过氧化相关的铁死亡性细胞死亡是治疗帕金森病的合理策略。我们在这里报告了 PTC-041 的特征,它是一种用于治疗帕金森病的抗铁死亡还原型脂氧合酶抑制剂。在这些研究中,PTC-041 可有效保护源自原发性帕金森病患者的成纤维细胞免受脂质过氧化和随后的铁死亡性细胞死亡,并预防原代大鼠神经元培养物中与铁死亡相关的神经元丢失和星形胶质细胞增生。此外,PTC-041 可预防铁死亡介导的α-突触核蛋白在体外聚集和亚硝基化,表明抗铁死亡脂氧合酶抑制剂在减轻帕金森病等突触核蛋白病的致病方面可能具有潜在作用。我们进一步发现,PTC-041 在体内可预防突触核蛋白病,表明 PTC-041 治疗 Line 61 转基因小鼠可防止α-突触核蛋白聚集和磷酸化,并预防相关的神经元和非神经元细胞死亡。最后,我们表明,PTC-041 可防止半帕金森大鼠模型中 6-羟多巴胺诱导的运动功能障碍,进一步验证了脂氧合酶抑制剂在治疗帕金森病中的潜在治疗益处。