转变血友病管理:来自基因治疗临床试验的经验教训。

IF 2.5 4区 生物学 Q3 BIOCHEMISTRY & MOLECULAR BIOLOGY
Md Sadique Hussain, Mudasir Maqbool, Mohammed M Arab, Amita Joshi Rana, Sumel Ashique, Yumna Khan, Vikas Jakhmola, Gaurav Gupta
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引用次数: 0

摘要

基因治疗标志着血友病治疗的革命性变革,提供了持续内源性凝血因子合成的可能性,并限制了对外部因子补充的需求。腺相关病毒(AAV)载体在B型血友病中的初步实验由于免疫反应和暂时翻译而受到限制。载体技术的进步,特别是通过自我互补的AAV创新和密码子优化的迷你因子IX (FIX)概念,显著改善了转导性能并延长了FIX活性。初步调查显示了令人鼓舞的结果,某些个体在8年以上的时间里保持一致的FIX表达;因此,减少每年出血事件和需要预防性治疗。由于因子VIII (FVIII)基因的巨大尺寸,血友病A基因治疗的发展遇到了实质性的障碍。最近使用编码b结构域缺失FVIII构建体的AAV血清型5 (AAV5)载体进行的实验显示,出血事件的发生率持续下降。研究表明FVIII表达时间延长,一些个体的凝血效率几乎达到正常水平。III期研究证实了长期的有效性和安全性,短暂的转氨酶升高是最常见的不良事件。尽管取得了这些进展,但困难仍然存在,包括对载体衣壳的免疫反应、肝毒性和翻译水平的不可预测性。包括慢病毒载体、基因编辑技术和新型定制连接策略在内的创新方法展示了增强基因治疗有效性的可能性。持续的临床研究和改进的输送系统将是至关重要的,以证实基因治疗作为血友病的最终途径。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Transforming Hemophilia Management: Lessons from Gene Therapy Clinical Trials.

Gene therapy signifies a transformative revolution in hemophilia care, providing the possibility for sustained endogenous synthesis of coagulation factors and limiting the need for external factor supplementation. Preliminary experiments in hemophilia B via adeno-associated viral (AAV) vectors encountered constraints owing to immunological reactions and temporary translation. Progress in vector technology, particularly via self-complementary AAV innovation and codon-optimized mini-factor IX (FIX) concepts, has markedly improved transduction performance and prolonged FIX activity. Initial investigations have shown encouraging outcomes, with certain individuals sustaining consistent FIX expressions for more than 8 years; hence, decreasing yearly bleeding incidents and requiring preventive therapy. The development of gene therapy for hemophilia A has encountered substantial obstacles owing to the enormous size of the factor VIII (FVIII) gene. The recent experiments using AAV serotypes 5 (AAV5) vectors that encode B-domain-deleted FVIII constructs have shown sustained levels along with substantial decreases in hemorrhage incidents. Research has shown prolonged FVIII expression, with some individuals attaining almost normal coagulation efficiency. Phase III studies have validated long-term effectiveness and safety, with transient transaminase elevations being the most common adverse event. Notwithstanding these advancements, difficulties persist, including immunological reactions to vector capsids, hepatotoxicity, and unpredictability in translation levels. Innovative approaches including lentiviral vectors, gene-editing technologies, and novel customized connection strategies demonstrate possibilities for enhancing the effectiveness of gene therapy. Continuous clinical research and improvement in delivery systems will be crucial in substantiating gene therapy as a definitive approach for hemophilia.

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来源期刊
Molecular Biotechnology
Molecular Biotechnology 医学-生化与分子生物学
CiteScore
4.10
自引率
3.80%
发文量
165
审稿时长
6 months
期刊介绍: Molecular Biotechnology publishes original research papers on the application of molecular biology to both basic and applied research in the field of biotechnology. Particular areas of interest include the following: stability and expression of cloned gene products, cell transformation, gene cloning systems and the production of recombinant proteins, protein purification and analysis, transgenic species, developmental biology, mutation analysis, the applications of DNA fingerprinting, RNA interference, and PCR technology, microarray technology, proteomics, mass spectrometry, bioinformatics, plant molecular biology, microbial genetics, gene probes and the diagnosis of disease, pharmaceutical and health care products, therapeutic agents, vaccines, gene targeting, gene therapy, stem cell technology and tissue engineering, antisense technology, protein engineering and enzyme technology, monoclonal antibodies, glycobiology and glycomics, and agricultural biotechnology.
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