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从逐个疾病转移:新型平台技术的筛查、试验设计和监管影响。

Moving away from one disease at a time: Screening, trial design, and regulatory implications of novel platform technologies.

机构信息

Takeda, Cambridge, Massachusetts, USA.

Division of Rare Diseases Research Innovation, National Center for Advancing Translational Sciences, National Institutes of Health, Bethesda, Maryland, USA.

出版信息

Am J Med Genet C Semin Med Genet. 2023 Mar;193(1):30-43. doi: 10.1002/ajmg.c.32031. Epub 2023 Feb 4.

Abstract

Most rare diseases are caused by single-gene mutations, and as such, lend themselves to a host of new gene-targeted therapies and technologies including antisense oligonucleotides, phosphomorpholinos, small interfering RNAs, and a variety of gene delivery and gene editing systems. Early successes are encouraging, however, given the substantial number of distinct rare diseases, the ability to scale these successes will be unsustainable without new development efficiencies. Herein, we discuss the need for genomic newborn screening to match pace with the growing development of targeted therapeutics and ability to rapidly develop individualized therapies for rare variants. We offer approaches to move beyond conventional "one disease at a time" preclinical and clinical drug development and discuss planned regulatory innovations that are necessary to speed therapy delivery to individuals in need. These proposals leverage the shared properties of platform classes of therapeutics and innovative trial designs including master and platform protocols to better serve patients and accelerate drug development. Ultimately, there are risks to these novel approaches; however, we believe that close partnership and transparency between health authorities, patients, researchers, and drug developers present the path forward to overcome these challenges and deliver on the promise of gene-targeted therapies for rare diseases.

摘要

大多数罕见病是由单基因突变引起的,因此,它们适用于许多新的基因靶向治疗和技术,包括反义寡核苷酸、磷酸吗啉代寡核苷酸、小干扰 RNA 以及各种基因传递和基因编辑系统。早期的成功令人鼓舞,然而,鉴于大量不同的罕见疾病,要想在没有新的开发效率的情况下扩大这些成功,将是不可持续的。在此,我们讨论了基因组新生儿筛查需要与靶向治疗的不断发展以及为罕见变异快速开发个体化治疗的能力相匹配的必要性。我们提出了超越传统的“一次一种疾病”的临床前和临床药物开发方法,并讨论了计划中的监管创新,这些创新对于加快向有需要的个人提供治疗是必要的。这些建议利用了治疗平台类药物和创新试验设计的共同特性,包括主方案和平台方案,以更好地为患者服务并加速药物开发。最终,这些新方法存在风险;然而,我们相信,卫生当局、患者、研究人员和药物开发者之间的密切合作和透明度为克服这些挑战并实现基因靶向治疗罕见病的承诺提供了前进的道路。

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