John van Geest Centre for Brain Repair, University of Cambridge, Cambridge CB2 0PY, UK; MRC Mitochondrial Biology Unit, Department of Clinical Neurosciences, University of Cambridge, Cambridge CB2 0XY, UK; Cambridge Eye Unit, Addenbrooke's Hospital, Cambridge University Hospitals NHS Foundation Trust, Cambridge CB2 0QQ, UK; Moorfields Eye Hospital NHS Foundation Trust, London EC1V 2PD, UK; Institute of Ophthalmology, University College London, London EC1V 9EL, UK.
IRCCS Istituto di Scienze Neurologiche di Bologna, Programma di Neurogenetica, 40139 Bologna, Italy; Department of Biomedical and Neuromotor Sciences, University of Bologna, 40127 Bologna, Italy.
Cell Rep Med. 2024 Mar 19;5(3):101437. doi: 10.1016/j.xcrm.2024.101437. Epub 2024 Feb 29.
Leber hereditary optic neuropathy (LHON) is a mitochondrial disease leading to rapid and severe bilateral vision loss. Idebenone has been shown to be effective in stabilizing and restoring vision in patients treated within 1 year of onset of vision loss. The open-label, international, multicenter, natural history-controlled LEROS study (ClinicalTrials.gov NCT02774005) assesses the efficacy and safety of idebenone treatment (900 mg/day) in patients with LHON up to 5 years after symptom onset (N = 199) and over a treatment period of 24 months, compared to an external natural history control cohort (N = 372), matched by time since symptom onset. LEROS meets its primary endpoint and confirms the long-term efficacy of idebenone in the subacute/dynamic and chronic phases; the treatment effect varies depending on disease phase and the causative mtDNA mutation. The findings of the LEROS study will help guide the clinical management of patients with LHON.
Leber 遗传性视神经病变(LHON)是一种线粒体疾病,可导致迅速而严重的双侧视力丧失。已有研究表明,依地苯醌可有效稳定和恢复发病后 1 年内接受治疗的患者的视力。开放性、国际、多中心、自然史对照的 LEROS 研究(ClinicalTrials.gov NCT02774005)评估了依地苯醌治疗(900mg/天)在症状发作后 5 年内的患者(N=199)和 24 个月的治疗期间的疗效和安全性,与通过症状发作时间匹配的外部自然史对照队列(N=372)进行比较。LEROS 达到了主要终点,并证实了依地苯醌在亚急性/动态期和慢性期的长期疗效;治疗效果取决于疾病阶段和致病 mtDNA 突变。LEROS 研究的结果将有助于指导 LHON 患者的临床管理。