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法齐西兰治疗α-1抗胰蛋白酶缺乏相关肝病——SEQUOIA 2期试验结果

Fazirsiran for the treatment of alpha-1 antitrypsin deficiency-associated liver disease findings from the SEQUOIA phase 2 trial.

作者信息

Liaquat Talha, Malik Bareera Tanveer, Hashmi Tayyab Habib, Arain Huzaifa, Ali Mohammad Haris, Haque Md Ariful

机构信息

Department of Medicine, Shaikh Khalifa Bin Zayed Al-Nahyan Medical College, Lahore, Pakistan.

Voice of Doctors Research School, Dhaka, Bangladesh.

出版信息

Ann Med Surg (Lond). 2025 May 30;87(7):4014-4016. doi: 10.1097/MS9.0000000000003393. eCollection 2025 Jul.

DOI:10.1097/MS9.0000000000003393
PMID:40851970
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12369710/
Abstract

Alpha-1 antitrypsin deficiency (AATD) is a prevalent autosomal recessive disorder affecting approximately 3 million people worldwide, caused by mutations in the SERPINA1 gene leading to low alpha-1 antitrypsin (AAT) levels. This deficiency predisposes individuals to chronic obstructive pulmonary disease, bronchiectasis, neonatal cholestasis, and liver cirrhosis. The most common pathogenic mutation, PI*ZZ allele, produces misfolded Z-AAT protein accumulating in hepatocytes. Currently, there are no specific treatments for AATD-related liver disease, with management focusing on preventing complications. Fazirsiran, an investigational RNA interference therapeutic, targets hepatocytes to reduce Z-AAT synthesis. Preclinical studies and an open-label phase 2 trial showed promising results. The subsequent phase 2 SEQUOIA trial, a randomized controlled study, evaluated Fazirsiran's efficacy and safety in 40 PiZZ homozygous patients. Results demonstrated significant dose-dependent reductions in serum Z-AAT levels (-61% to -94%) and liver Z-AAT concentrations, improved liver histopathology, and a favorable safety profile. These findings support Fazirsiran's potential as a therapeutic option for AATD-associated liver disease.

摘要

α-1抗胰蛋白酶缺乏症(AATD)是一种常见的常染色体隐性疾病,全球约有300万人受其影响,由SERPINA1基因突变导致α-1抗胰蛋白酶(AAT)水平降低引起。这种缺乏使个体易患慢性阻塞性肺疾病、支气管扩张、新生儿胆汁淤积和肝硬化。最常见的致病突变PI*ZZ等位基因会产生错误折叠的Z-AAT蛋白,在肝细胞中积累。目前,尚无针对AATD相关肝病的特异性治疗方法,治疗重点在于预防并发症。Fazirsiran是一种正在研究的RNA干扰疗法,作用于肝细胞以减少Z-AAT的合成。临床前研究和一项开放标签的2期试验显示了有前景的结果。随后的2期SEQUOIA试验是一项随机对照研究,评估了Fazirsiran在40名PIZZ纯合患者中的疗效和安全性。结果显示血清Z-AAT水平(-61%至-94%)和肝脏Z-AAT浓度显著降低,肝脏组织病理学得到改善,且安全性良好。这些发现支持Fazirsiran作为AATD相关肝病治疗选择的潜力。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c0f0/12369710/1199cdcbf93b/ms9-87-4014-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c0f0/12369710/1199cdcbf93b/ms9-87-4014-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c0f0/12369710/1199cdcbf93b/ms9-87-4014-g001.jpg

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本文引用的文献

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Fazirsiran for Adults With Alpha-1 Antitrypsin Deficiency Liver Disease: A Phase 2 Placebo Controlled Trial (SEQUOIA).法齐里斯兰治疗成人 α-1 抗胰蛋白酶缺乏症肝病的 2 期安慰剂对照试验(SEQUOIA)
Gastroenterology. 2024 Oct;167(5):1008-1018.e5. doi: 10.1053/j.gastro.2024.06.028. Epub 2024 Jul 2.
2
RNAi therapeutics for diseases involving protein aggregation: fazirsiran for alpha-1 antitrypsin deficiency-associated liver disease.用于治疗涉及蛋白质聚集疾病的RNA干扰疗法:法齐司兰用于治疗α-1抗胰蛋白酶缺乏症相关肝病。
Expert Opin Investig Drugs. 2023 Jul-Dec;32(7):571-581. doi: 10.1080/13543784.2023.2239707. Epub 2023 Jul 24.
3
Fazirsiran for Liver Disease Associated with Alpha-Antitrypsin Deficiency.
法齐里斯兰治疗 α1-抗胰蛋白酶缺乏症相关肝病。
N Engl J Med. 2022 Aug 11;387(6):514-524. doi: 10.1056/NEJMoa2205416. Epub 2022 Jun 25.
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Aliment Pharmacol Ther. 2018 Apr;47(7):877-885. doi: 10.1111/apt.14537. Epub 2018 Feb 15.
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Liver disease in alpha-1 antitrypsin deficiency: current understanding and future therapy.α-1 抗胰蛋白酶缺乏症相关肝病:当前认识与未来治疗。
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Alpha-1 antitrypsin deficiency is not a rare disease but a disease that is rarely diagnosed.α-1抗胰蛋白酶缺乏症并非罕见疾病,而是一种很少被诊断出来的疾病。
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The mechanism of Z alpha 1-antitrypsin accumulation in the liver.Zα1-抗胰蛋白酶在肝脏中积聚的机制。
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