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儿童脊髓性肌萎缩症。

Childhood spinal muscular atrophy.

机构信息

Department of Clinical Medicine and Neuroscience, CUNY School of Medicine, New York, NY, United States; Department of Medicine, Section of Internal Medicine and Neurology, White Plains Hospital, White Plains, NY, United States.

Department of Neurology and Pediatrics, Center for Gene Therapy, Abigail Wexner Research Institute, The Ohio State University, Nationwide Children's Hospital, Columbus, OH, United States.

出版信息

Handb Clin Neurol. 2023;196:43-58. doi: 10.1016/B978-0-323-98817-9.00030-2.

Abstract

Spinal muscular atrophy (SMA) is caused by biallelic mutations in the SMN1 (survival motor neuron 1) gene on chromosome 5q13.2, which leads to a progressive degeneration of alpha motor neurons in the spinal cord and in motor nerve nuclei in the caudal brainstem. It is characterized by progressive proximally accentuated muscle weakness with loss of already acquired motor skills, areflexia and, depending on the phenotype, varying degrees of weakness of the respiratory and bulbar muscles. Over the past decade, disease-modifying therapies have become available based on splicing modulation of the SMN2 with SMN1 gene replacement, which if initiated significantly modifies the natural course of the disease. Newborn screening for SMA has been implemented in an increasing number of centers; however, available evidence for these new treatments is often limited to a small spectrum of patients concerning age and disease stage.

摘要

脊髓性肌萎缩症(SMA)是由 5q13.2 染色体上的 SMN1(生存运动神经元 1)基因的双等位基因突变引起的,导致脊髓中的α运动神经元和尾部脑干中的运动神经核进行性退化。其特征是进行性近端肌肉无力,已获得的运动技能丧失,反射消失,并且根据表型的不同,呼吸肌和延髓肌的无力程度也不同。在过去的十年中,已经出现了基于 SMN2 剪接调节和 SMN1 基因替代的疾病修饰疗法,如果早期开始治疗,会显著改变疾病的自然进程。越来越多的中心已经开展了 SMA 的新生儿筛查;然而,这些新疗法的现有证据通常仅限于年龄和疾病阶段的一小部分患者。

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