使用帕西兰治疗遗传性转甲状腺素介导淀粉样变性多神经病变的实用指南。

IF 2.8 3区 医学 Q1 Pharmacology, Toxicology and Pharmaceutics
Therapeutics and Clinical Risk Management Pub Date : 2023-11-27 eCollection Date: 2023-01-01 DOI:10.2147/TCRM.S361706
Stacy Dixon, Xuan Kang, Dianna Quan
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引用次数: 0

摘要

变异型甲状腺转蛋白淀粉样变性(ATTRv)是一种常染色体显性遗传性遗传病,全世界有5000- 10000人患病。 它是由转甲状腺素(TTR)基因突变引起的,由于TTR蛋白原纤维的异常积累,导致多种器官的淀粉样蛋白沉积。 虽然这是一种多系统疾病,但心脏和周围神经是优先受影响的器官。 超过150个TTR基因突变与这种疾病相关,临床表型可能差异很大。 严重的这种疾病可能是致命的。 幸运的是,基于寡核苷酸的治疗时代已经导致了几种新的治疗选择的发展。 Patisiran是一种封装在脂质纳米颗粒中的小干扰RNA (siRNA),可靶向突变型和野生型TTR,并导致血清和组织沉积物中TTR蛋白的显著减少。 Patisiran已在美国和欧盟被批准用于治疗遗传性trr介导淀粉样变性引起的成人多发性神经病变。 在这篇综述中,我们将讨论patisiran的发展,导致治疗批准的临床试验,并提供用于临床实践的指导参数  。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Practical Guidance for the Use of Patisiran in the Management of Polyneuropathy in Hereditary Transthyretin-Mediated Amyloidosis.

Variant transthyretin amyloidosis (ATTRv) is an autosomal dominant inherited genetic disorder that affects 5000-10,000 people worldwide. It is caused by mutations in the transthyretin (TTR) gene and results in amyloid deposition in a variety of organs due to abnormal accumulation of TTR protein fibrils. Although this is a multisystem disorder, the heart and peripheral nerves are the preferentially affected organs. Over 150 TTR gene mutations have been associated with this disease and the clinical phenotype can vary significantly. Severe forms of the disorder can be fatal. Fortunately, the oligonucleotide-based therapy era has resulted in the development of several novel treatment options. Patisiran is a small interfering RNA (siRNA) encapsulated in a lipid nanoparticle that targets both mutant and wild-type TTR and results in significant reductions of the TTR protein in the serum and in tissue deposits. Patisiran has been approved for treatment of adults with polyneuropathy due to hereditary TTR-mediated amyloidosis in both the United States (US) and European Union (EU). In this review, we will discuss the development of patisiran, the clinical trials that lead to treatment approval, and provide guideline parameters for use in clinical practice.  .

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来源期刊
Therapeutics and Clinical Risk Management
Therapeutics and Clinical Risk Management HEALTH CARE SCIENCES & SERVICES-
CiteScore
5.30
自引率
3.60%
发文量
139
审稿时长
16 weeks
期刊介绍: Therapeutics and Clinical Risk Management is an international, peer-reviewed journal of clinical therapeutics and risk management, focusing on concise rapid reporting of clinical studies in all therapeutic areas, outcomes, safety, and programs for the effective, safe, and sustained use of medicines, therapeutic and surgical interventions in all clinical areas. The journal welcomes submissions covering original research, clinical and epidemiological studies, reviews, guidelines, expert opinion and commentary. The journal will consider case reports but only if they make a valuable and original contribution to the literature. As of 18th March 2019, Therapeutics and Clinical Risk Management will no longer consider meta-analyses for publication. The journal does not accept study protocols, animal-based or cell line-based studies.
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