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由复合杂合 SMN1 突变引起的脊髓性肌萎缩症:两例病例报告及文献复习。

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

机构信息

Department of Neurology, Shengjing Hospital of China Medical University, Shenyang, Liaoning, 110000, China.

出版信息

Neurol Sci. 2024 Dec;45(12):5605-5615. doi: 10.1007/s10072-024-07651-0. Epub 2024 Jul 8.

Abstract

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.

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退化,导致对称性肌肉无力和萎缩。描述了两种新型 SMN1 突变(患者 1:c.683T > A,p.Leu228Ter;患者 2:c.347 T > C,p.Ile116 Thr)。我们报道了两名具有 SMN1 突变的 SMA 患者的临床特征,并对之前报道的 22 例病例进行了文献复习。两名 SMA 患者表现为进行性近端下肢无力和较轻的临床症状。在总共 22 例病例中,最常见的 SMN1 基因突变是错义突变(55%),其次是剪接缺陷(27%)、无义(9%)和移码(9%)。我们讨论了这些基因内突变在表型结果中可能起决定性作用,丰富了 SMN1 精细突变数据库。

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