脊髓性肌萎缩症的最新进展。

IF 2.5 3区 医学 Q1 MEDICINE, GENERAL & INTERNAL
Wen-Chin Weng, Wang-Tso Lee, Yin-Hsiu Chien, Li-Kai Tsai
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引用次数: 0

摘要

脊髓性肌萎缩症(SMA)是一种由SMN1基因双等位致病变异引起的进行性神经肌肉疾病,其疾病严重程度主要由SMN2基因拷贝数修饰。在过去十年中,随着三种疾病改善疗法的批准,SMA治疗取得了显着进展:nusinersen,一种反义寡核苷酸;Onasemnogene abeparvovec,一种基因替代疗法;还有risdiplam,一种口腔剪接修饰剂。这些疗法已经将SMA从一种致命疾病转变为一种可治疗的疾病。临床试验和现实世界的经验证实了它们对症状前、婴儿发病和晚发病患者的疗效,尽管开始治疗的年龄、治疗前疾病持续时间、基线功能和SMN2拷贝影响结果。正在进行的研究正在探索优化剂量,联合治疗和smn独立策略,如肌生长抑制素抑制和神经肌肉连接增强。同时,新生儿筛查的出现使症状前干预成为可能,尽管在确定特定基因型的最佳时机和管理具有4个SMN2拷贝的患者方面仍然存在挑战。包括复合肌肉动作电位和循环神经丝在内的生物标志物正在评估中,以完善个性化治疗策略。随着生存率的提高,新的表型和多系统表现正在出现,强调需要综合多学科治疗和更新指南。从SMA治疗发展中吸取的经验教训可以作为其他神经系统疾病的范例。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Updates of spinal muscular atrophy in advanced therapies.

Spinal muscular atrophy (SMA) is a progressive neuromuscular disorder caused by biallelic pathogenic variants in SMN1 gene, with disease severity mainly modified by SMN2 gene copies. There are remarkable advances in SMA treatment in the last decade with the approval of three disease-modifying therapies: nusinersen, an antisense oligonucleotide; onasemnogene abeparvovec, a gene replacement therapy; and risdiplam, an oral splicing modifier. These therapies have transformed SMA from a fatal condition into a treatable disease. Clinical trials and real-world experiences have confirmed their efficacy across presymptomatic, infantile-onset, and later-onset patients, though age at treatment initiation, pre-treatment disease duration, baseline function, and SMN2 copies influence outcomes. Ongoing research is exploring optimized dosing, combination therapies, and SMN-independent strategies, such as myostatin inhibition and neuromuscular junction enhancement. Meanwhile, the advent of newborn screening enables presymptomatic intervention, though challenges remain in identifying optimal timing for specific genotypes and managing patients with 4 SMN2 copies. Biomarkers including compound muscle action potential and circulating neurofilaments are under evaluation to refine personalized treatment strategies. As survival improves, new phenotypes and multisystem manifestations are emerging, underscoring the need for integrated multidisciplinary care and updated guidelines. Lessons learned from SMA therapy development may serve as a paradigm for other neurological diseases.

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来源期刊
CiteScore
6.50
自引率
6.20%
发文量
381
审稿时长
57 days
期刊介绍: Journal of the Formosan Medical Association (JFMA), published continuously since 1902, is an open access international general medical journal of the Formosan Medical Association based in Taipei, Taiwan. It is indexed in Current Contents/ Clinical Medicine, Medline, ciSearch, CAB Abstracts, Embase, SIIC Data Bases, Research Alert, BIOSIS, Biological Abstracts, Scopus and ScienceDirect. As a general medical journal, research related to clinical practice and research in all fields of medicine and related disciplines are considered for publication. Article types considered include perspectives, reviews, original papers, case reports, brief communications, correspondence and letters to the editor.
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