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携带SOD1 p.Gly13Arg突变患者的散发性肌萎缩侧索硬化症快速进展

Rapid Progression of Sporadic ALS in a Patient Carrying SOD1 p.Gly13Arg Mutation.

作者信息

Kim Myung-Jin, Bae Jae-Han, Kim Jeong-Min, Kim Hye Ryoun, Yoon Byung-Nam, Sung Jung-Joon, Ahn Suk-Won

机构信息

Department of Neurology, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul 06973, Korea.

Department of Laboratory Medicine, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul 06973, Korea.

出版信息

Exp Neurobiol. 2016 Dec;25(6):347-350. doi: 10.5607/en.2016.25.6.347. Epub 2016 Dec 15.

DOI:10.5607/en.2016.25.6.347
PMID:28035186
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5195821/
Abstract

Amyotrophic lateral sclerosis (ALS), the most common adult onset motor neuron disease, is pathologically characterized by progressive loss of the upper and lower motor neurons. Mutations in the Cu/Zn superoxide dismutase gene (SOD1) account for about 20% of familial ALS cases and a small percentage of sporadic ALS (SALS) cases, and have revealed a validated genotype-phenotype correlation. Herein, we report a p.Gly13Arg mutation in SOD1 exon 1 in a patient with SALS who presented with a rapidly progressive course, predominantly affecting the lower motor neurons. A 48-year-old man presented with progressive weakness and muscle atrophy of the left upper and lower limbs, followed by muscle fasciculation and cramping. The clinical features of the patient were clearly suggestive of ALS, and implied a sporadic form with rapid progression, predominantly affecting the lower motor neurons. Sequencing of the SOD1 gene by PCR revealed a missense mutation of G to C (c.37G>C) in exon 1, and amino acid substitution of glycine by arginine (p.Gly13Arg). This is the first case identifying the p.Gly13Arg mutation of SOD1 in the Korean population, and clinical assessments of this patient revealed a different phenotype compared with other cases.

摘要

肌萎缩侧索硬化症(ALS)是最常见的成人起病的运动神经元疾病,其病理特征是上下运动神经元进行性丧失。铜/锌超氧化物歧化酶基因(SOD1)突变约占家族性ALS病例的20%,以及一小部分散发性ALS(SALS)病例,并揭示了一种经过验证的基因型-表型相关性。在此,我们报告一例SALS患者,其SOD1外显子1存在p.Gly13Arg突变,病程进展迅速,主要影响下运动神经元。一名48岁男性出现左上肢和下肢进行性无力和肌肉萎缩,随后出现肌肉束颤和痉挛。该患者的临床特征明显提示为ALS,提示为散发性形式,进展迅速,主要影响下运动神经元。通过PCR对SOD1基因进行测序,发现外显子1存在G到C的错义突变(c.37G>C),以及甘氨酸被精氨酸取代(p.Gly13Arg)。这是韩国人群中首例鉴定出SOD1的p.Gly13Arg突变的病例,对该患者的临床评估显示其表型与其他病例不同。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b85/5195821/35521ceabeb4/en-25-347-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b85/5195821/35521ceabeb4/en-25-347-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b85/5195821/35521ceabeb4/en-25-347-g001.jpg

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本文引用的文献

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Identification of mutations in Korean patients with amyotrophic lateral sclerosis using multigene panel testing.使用多基因检测面板鉴定韩国肌萎缩侧索硬化症患者的突变
Neurobiol Aging. 2016 Jan;37:209.e9-209.e16. doi: 10.1016/j.neurobiolaging.2015.09.012. Epub 2015 Sep 30.
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Comparison between Flail Arm Syndrome and Upper Limb Onset Amyotrophic Lateral Sclerosis: Clinical Features and Electromyographic Findings.
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Exp Neurobiol. 2014 Sep;23(3):253-7. doi: 10.5607/en.2014.23.3.253. Epub 2014 Sep 18.
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Amyotrophic lateral sclerosis - cell based therapy and novel therapeutic development.肌萎缩侧索硬化症——基于细胞的疗法与新型治疗方法的开发
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Slowly progressing lower motor neuron disease caused by a novel duplication mutation in exon 1 of the SOD1 gene.
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Screening of the SOD1, FUS, TARDBP, ANG, and OPTN mutations in Korean patients with familial and sporadic ALS.对韩国家族性和散发性 ALS 患者的 SOD1、FUS、TARDBP、ANG 和 OPTN 突变进行筛查。
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