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脊髓性肌萎缩症的治疗进展。

Therapeutic developments in spinal muscular atrophy.

机构信息

Division of Pediatric Neurosciences, Department of Neurology, SMA Clinical Research Center, Columbia University Medical Center, Harkness Pavilion, HP-514, 180 Fort Washington Avenue, New York, NY 10032-3791, USA.

出版信息

Ther Adv Neurol Disord. 2010 May;3(3):173-85. doi: 10.1177/1756285610369026.

Abstract

Spinal muscular atrophy (SMA), a potentially devastating disease marked by progressive weakness and muscle atrophy resulting from the dysfunction and loss of motor neurons of the spinal cord, has emerged in recent years as an attractive target for therapeutic intervention. Caused by a homozygous mutation to the Survival of Motor Neurons 1 (SMN1) gene on chromosome 5q, the severity of the clinical phenotype in SMA is modulated by the function of a related protein, Survival of Motor Neurons 2 (SMN2). SMN2 predominantly produces an unstable SMN transcript lacking exon 7; only about 10% of the transcription product produces a full-length, functional SMN protein. Several therapeutic strategies have targeted this gene with the goal of producing increased full-length SMN transcript, thereby modifying the underlying mechanism. Drugs that have increased SMN2 function, in vitro, are now explored for potential therapeutic benefit in this disease. Alternative approaches, including neuroprotective, muscle anabolic, gene and cell replacement strategies, also hold promise. The recent advances in preclinical research and the development of a wider range of animal models for SMA continue to provide cautious optimism that effective treatments for SMA will eventually emerge.

摘要

脊髓性肌萎缩症(SMA)是一种潜在的破坏性疾病,其特征是脊髓运动神经元功能障碍和丧失,导致进行性肌无力和肌肉萎缩。近年来,它已成为治疗干预的一个有吸引力的靶点。SMA 是由 5q 染色体上运动神经元生存基因 1(SMN1)的纯合突变引起的,其临床表型的严重程度受相关蛋白运动神经元生存基因 2(SMN2)的功能调节。SMN2 主要产生缺乏外显子 7 的不稳定 SMN 转录本;只有大约 10%的转录产物产生全长、功能性 SMN 蛋白。几种治疗策略都以该基因为靶点,旨在产生更多的全长 SMN 转录本,从而改变潜在的发病机制。目前正在研究体外增加 SMN2 功能的药物,以评估其在该疾病中的潜在治疗效果。其他方法,包括神经保护、肌肉合成代谢、基因和细胞替代策略,也有希望。最近在 SMA 的临床前研究和更广泛的动物模型开发方面的进展,继续为 SMA 的有效治疗方法最终出现提供了谨慎的乐观态度。

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