Goldberg Yigal Paul, Navon-Perry Leehee, Cruz-Herranz Andrés, Chen Kelly, Hecker-Barth Gabriele, Spiegel Katrin, Cohen Yael, Niethammer Martin, Tan Andrew M, Schuring Henk, Geva Michal, Hayden Michael R
Prilenia Therapeutics B.V., Naarden, The Netherlands.
GHB Hecker-Barth Biopharma Consulting GmbH, Konstanz, Germany.
CNS Drugs. 2025 May;39(5):485-498. doi: 10.1007/s40263-025-01171-x. Epub 2025 Mar 7.
Huntington's disease (HD) is a rare, fatal, chronic progressive neurodegenerative disorder with a significant unmet medical need for effective treatments. Pridopidine is a novel, first-in-class, highly selective and potent sigma-1 receptor (S1R) agonist in development for HD. Pridopidine has been extensively studied in adult HD across the full spectrum of disease severity and age ranges, and its safety profile has been characterized in approximately 1600 participants across multiple studies and a broad range of doses. The specific objective of this study was to gain an in-depth understanding of pridopidine's safety profile at the recommended human dose of 45 mg twice daily (bid) in patients with HD.
An integrated safety analysis of pooled data from 1067 patients with HD enrolled in four double-blind, placebo-controlled studies was performed. The safety profile of pridopidine was compared with placebo.
Pridopidine was found to be generally safe and well tolerated with an adverse event (AE) profile comparable to that of placebo. Moreover, there were no significant differences observed in the safety profile of pridopidine compared with placebo when analyzed by age, sex, baseline total functional capacity (TFC), cytosine-adenine-guanine (CAG) repeat length, use of antidopaminergic medications (ADMs), and region.
The integrated analysis replicated and corroborated the good safety profile observed in the individual studies. Despite the larger sample size, no new safety signals emerged. Long-term exposure to pridopidine, up to 6.5 years in open-label extension studies, revealed no new safety concerns, supporting its potential for long-term use in patients with HD.
亨廷顿病(HD)是一种罕见的、致命的慢性进行性神经退行性疾病,对有效治疗存在重大未满足的医疗需求。普立哌啶是一种新型的、同类首创的、高选择性且强效的σ-1受体(S1R)激动剂,正在研发用于治疗HD。普立哌啶已在成年HD患者中针对疾病严重程度和年龄范围的全谱进行了广泛研究,并且在多项研究中的约1600名参与者以及广泛的剂量范围内对其安全性进行了表征。本研究的具体目标是深入了解普立哌啶在HD患者中每日两次45 mg推荐人用剂量下的安全性。
对纳入四项双盲、安慰剂对照研究的1067例HD患者的汇总数据进行了综合安全性分析。将普立哌啶的安全性与安慰剂进行了比较。
发现普立哌啶总体安全且耐受性良好,不良事件(AE)谱与安慰剂相当。此外,按年龄、性别、基线总功能能力(TFC)、胞嘧啶-腺嘌呤-鸟嘌呤(CAG)重复长度、抗多巴胺能药物(ADM)的使用情况和地区进行分析时,普立哌啶与安慰剂相比,安全性方面未观察到显著差异。
综合分析重复并证实了在各项个体研究中观察到的良好安全性。尽管样本量更大,但未出现新的安全信号。在开放标签扩展研究中长达6.5年的普立哌啶长期暴露未显示新的安全问题,支持其在HD患者中长期使用的潜力。