SMN1复合杂合子突变导致的脊髓性肌肉萎缩症:两个病例和文献综述。

IF 2.7 4区 医学 Q2 CLINICAL NEUROLOGY
Neurological Sciences Pub Date : 2024-12-01 Epub Date: 2024-07-08 DOI:10.1007/s10072-024-07651-0
Yuewei Chi, Yue Qiao, Ying Ma
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引用次数: 0

摘要

脊髓性肌萎缩症(SMA)是一种罕见的神经肌肉疾病,其特征是运动神经元变性,导致对称性肌肉无力和萎缩。描述两个新型 SMN1 突变(患者 1:c.683T > A,p.Leu228Ter;患者 2:c.347 T > C,p.Ile116 Thr)。我们报告了两例SMN1突变患者的临床特征,并对之前报告的22例病例进行了文献回顾。两名SMA患者表现为进行性下肢近端无力,临床症状较轻。在总共22例病例中,最常见的SMN1基因改变是错义突变(55%),其次是剪接缺陷(27%)、无义突变(9%)和框架移位(9%)。我们讨论了这些基因内突变在表型结果中可能起到的决定性作用,这些突变丰富了 SMN1 基因精细突变数据库。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Spinal muscular atrophy caused by compound heterozygous SMN1 mutations: two cases and literature review.

Spinal muscular atrophy caused by compound heterozygous SMN1 mutations: two cases and literature review.

Spinal muscular atrophy (SMA) is a rare neuromuscular disease, which is characterized by the degeneration of motor neurons, leading to symmetrical muscle weakness and atrophy. Description of two novel SMN1 mutations (patient1: c.683T > A, p.Leu228Ter; patient2: c.347 T > C, p.Ile116 Thr). We reported two patients with SMN1 mutations with the clinical features, and provided a literature review of the previously reported 22 cases. Two SMA patients showed progressive proximal lower limb weakness and milder clinical symptom. In a total of 22 cases, the most commonly observed SMN1 gene alteration was missense mutation (55%), followed by splicing defect (27%), nonsense (9%) and frameshift (9%). We discuss the possible decisive role of these intragenic mutations in the phenotypic results, which enriched the SMN 1 fine mutation database.

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来源期刊
Neurological Sciences
Neurological Sciences 医学-临床神经学
CiteScore
6.10
自引率
3.00%
发文量
743
审稿时长
4 months
期刊介绍: Neurological Sciences is intended to provide a medium for the communication of results and ideas in the field of neuroscience. The journal welcomes contributions in both the basic and clinical aspects of the neurosciences. The official language of the journal is English. Reports are published in the form of original articles, short communications, editorials, reviews and letters to the editor. Original articles present the results of experimental or clinical studies in the neurosciences, while short communications are succinct reports permitting the rapid publication of novel results. Original contributions may be submitted for the special sections History of Neurology, Health Care and Neurological Digressions - a forum for cultural topics related to the neurosciences. The journal also publishes correspondence book reviews, meeting reports and announcements.
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