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信使 RNA 作为一种个体化治疗方法:罕见代谢疾病的关键时刻。

Messenger RNA as a personalized therapy: The moment of truth for rare metabolic diseases.

机构信息

Hepatology Program, Center for Applied Medical Research (CIMA), University of Navarra, Pamplona, Spain; Navarra Institute for Health Research (IDISNA), Pamplona, Spain.

Hepatology Program, Center for Applied Medical Research (CIMA), University of Navarra, Pamplona, Spain.

出版信息

Int Rev Cell Mol Biol. 2022;372:55-96. doi: 10.1016/bs.ircmb.2022.03.005. Epub 2022 May 16.

Abstract

Inborn errors of metabolism (IEM) encompass a group of monogenic diseases affecting both pediatric and adult populations and currently lack effective treatments. Some IEM such as familial hypercholesterolemia or X-linked protoporphyria are caused by gain of function mutations, while others are characterized by an impaired protein function, causing a metabolic pathway blockage. Pathophysiology classification includes intoxication, storage and energy-related metabolic disorders. Factors specific to each disease trigger acute metabolic decompensations. IEM require prompt and effective care, since therapeutic delay has been associated with the development of fatal events including severe metabolic acidosis, hyperammonemia, cerebral edema, and death. Rapid expression of therapeutic proteins can be achieved hours after the administration of messenger RNAs (mRNA), representing an etiological solution for acute decompensations. mRNA-based therapy relies on modified RNAs with enhanced stability and translatability into therapeutic proteins. The proteins produced in the ribosomes can be targeted to specific intracellular compartments, the cell membrane, or be secreted. Non-immunogenic lipid nanoparticle formulations have been optimized to prevent RNA degradation and to allow safe repetitive administrations depending on the disease physiopathology and clinical status of the patients, thus, mRNA could be also an effective chronic treatment for IEM. Given that the liver plays a key role in most of metabolic pathways or can be used as bioreactor for excretable proteins, this review focuses on the preclinical and clinical evidence that supports the implementation of mRNA technology as a promising personalized strategy for liver metabolic disorders such as acute intermittent porphyria, ornithine transcarbamylase deficiency or glycogen storage disease.

摘要

先天性代谢缺陷(IEM)涵盖了一组影响儿科和成人人群的单基因疾病,目前缺乏有效的治疗方法。一些 IEM,如家族性高胆固醇血症或 X 连锁原卟啉症,是由功能获得性突变引起的,而其他 IEM 的特征是蛋白质功能受损,导致代谢途径受阻。病理生理学分类包括中毒、储存和与能量相关的代谢紊乱。每种疾病特有的因素会引发急性代谢失代偿。IEM 需要及时有效的护理,因为治疗延迟与致命事件的发展有关,包括严重代谢性酸中毒、高氨血症、脑水肿和死亡。信使 RNA(mRNA)给药后数小时内可以快速表达治疗性蛋白,这为急性失代偿提供了一种病因治疗方法。基于 mRNA 的治疗依赖于经过修饰的 RNA,其稳定性增强,并且可以翻译成治疗性蛋白。核糖体中产生的蛋白质可以靶向特定的细胞内区室、细胞膜或分泌。非免疫原性脂质纳米颗粒制剂已被优化,以防止 RNA 降解,并根据疾病的病理生理学和患者的临床状况允许安全重复给药,因此,mRNA 也可能是 IEM 的有效慢性治疗方法。鉴于肝脏在大多数代谢途径中发挥关键作用,或者可以用作可分泌蛋白质的生物反应器,因此本综述重点介绍了支持将 mRNA 技术作为一种有前途的个性化策略用于治疗急性间歇性卟啉症、鸟氨酸转氨甲酰酶缺乏症或糖原贮积症等肝脏代谢紊乱的临床前和临床证据。

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