常染色体隐性非突发性耳聋:AAV 基因疗法真的有机会吗?

IF 2.1 Q1 AUDIOLOGY & SPEECH-LANGUAGE PATHOLOGY Audiology Research Pub Date : 2024-02-22 DOI:10.3390/audiolres14020022
Davide Brotto, Marco Greggio, Cosimo De Filippis, Patrizia Trevisi
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引用次数: 0

摘要

感音神经性听力损失的病因在很大程度上受基因突变的影响,约 80% 的病例归因于遗传原因,只有 20% 归因于环境因素。迄今为止,人类已发现 100 多种非综合征性耳聋基因。在非综合征感音神经性听力损伤中,约 75%-85% 的病例为常染色体隐性遗传。近年来,针对内耳疾病的分子基因疗法取得了突破性进展,显示出良好的效果。实验研究表明,只需局部注射携带额外正常基因的腺相关病毒载体,或使用核酶修改基因组,就能改善听力。这些开创性的方法为潜在的治疗干预开辟了新的可能性。按照 PRISMA 标准,我们总结了在不同的 DFNB 耳聋动物模型的临床前开发阶段显示听力改善的 AAV 基因治疗实验,以及目前处于临床阶段的针对常染色体隐性非综合征性听力损失的 AAV 基因治疗计划。共发现并列出了 17 项临床前研究和 3 项临床研究。尽管障碍重重,但在听力损失基因治疗的道路上已经取得了重大突破,为患者提供新颖、有效的治疗提供了巨大的潜力。
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Autosomal Recessive Non-Syndromic Deafness: Is AAV Gene Therapy a Real Chance?

The etiology of sensorineural hearing loss is heavily influenced by genetic mutations, with approximately 80% of cases attributed to genetic causes and only 20% to environmental factors. Over 100 non-syndromic deafness genes have been identified in humans thus far. In non-syndromic sensorineural hearing impairment, around 75-85% of cases follow an autosomal recessive inheritance pattern. In recent years, groundbreaking advancements in molecular gene therapy for inner-ear disorders have shown promising results. Experimental studies have demonstrated improvements in hearing following a single local injection of adeno-associated virus-derived vectors carrying an additional normal gene or using ribozymes to modify the genome. These pioneering approaches have opened new possibilities for potential therapeutic interventions. Following the PRISMA criteria, we summarized the AAV gene therapy experiments showing hearing improvement in the preclinical phases of development in different animal models of DFNB deafness and the AAV gene therapy programs currently in clinical phases targeting autosomal recessive non syndromic hearing loss. A total of 17 preclinical studies and 3 clinical studies were found and listed. Despite the hurdles, there have been significant breakthroughs in the path of HL gene therapy, holding great potential for providing patients with novel and effective treatment.

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来源期刊
Audiology Research
Audiology Research AUDIOLOGY & SPEECH-LANGUAGE PATHOLOGY-
CiteScore
2.30
自引率
23.50%
发文量
56
审稿时长
11 weeks
期刊介绍: The mission of Audiology Research is to publish contemporary, ethical, clinically relevant scientific researches related to the basic science and clinical aspects of the auditory and vestibular system and diseases of the ear that can be used by clinicians, scientists and specialists to improve understanding and treatment of patients with audiological and neurotological disorders.
期刊最新文献
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