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塞尔维亚最大的三级中心中基因检测呈阳性的肌萎缩侧索硬化症患者的临床和认知特征比较。

Comparison of the clinical and cognitive features of genetically positive ALS patients from the largest tertiary center in Serbia.

作者信息

Marjanović Ivan V, Selak-Djokić Biljana, Perić Stojan, Janković Milena, Arsenijević Vladimir, Basta Ivana, Lavrnić Dragana, Stefanova Elka, Stević Zorica

机构信息

Neurology Clinic, Clinical Center of Serbia, School of Medicine, University in Belgrade, Dr Subotica 6, 11000, Belgrade, Serbia.

Neurology Clinic, Clinical Center of Serbia, Belgrade, Serbia.

出版信息

J Neurol. 2017 Jun;264(6):1091-1098. doi: 10.1007/s00415-017-8495-y. Epub 2017 Apr 25.

DOI:10.1007/s00415-017-8495-y
PMID:28444446
Abstract

Discovering novel mutations in C9orf72, FUS, ANG, and TDP-43 genes in ALS patients arises necessities for better clinical characterizations of these subjects. The aim is to determine clinical and cognitive profile of genetically positive Serbian ALS patients. 241 ALS patients were included in the study (17 familiar and 224 apparently sporadic). The following genes were analyzed: SOD1, C9orf72, ANG, FUS, and TDP-43. An extensive battery of classic neuropsychological tests was used in 27 ALS patients (22 SOD1 positive and 5 SOD1 negative) and 82 healthy controls (HCs). Overall 37 (15.4%) of 241 ALS patients carried mutations in tested genes-among 17 familiar ALS patients 16 (94.1%) were positive and among 224 apparently sporadic 21 (9.4%) had causative mutation. Mutations in SOD1 gene were the most common, representing 27 (73.0%) of all genetically positive ALS patients. The main clinical characteristics of SOD1 positive patients were: spinal onset in lower extremities, common sphincter and sensitive disturbances, and dysexecutive syndrome. Within SOD1 positive patients, we noticed somewhat earlier onset in patients with A145G, sensory and sphincter disturbances were dominant in patients with L144F, while D90A patients had significant sensory involvement. SOD1 negative group consisted of ten (27.0%) patients (six C9orf72, two ANG, one TDP-43, and one patient baring triple FUS, C9orf72 expansion, and ANG variants). Bulbar involvement and more extensive neuropsychological impairment (including executive, visuospatial, and memory difficulties) were the main features of SOD1 negative cohort. Our results suggest that meaningful clinical suspicion of certain ALS genotype might be made based on thorough clinical evaluation of patients.

摘要

在肌萎缩侧索硬化症(ALS)患者中发现C9orf72、FUS、ANG和TDP - 43基因的新突变,使得对这些患者进行更好的临床特征描述成为必要。目的是确定基因检测呈阳性的塞尔维亚ALS患者的临床和认知特征。241例ALS患者纳入研究(17例家族性和224例明显散发性)。分析了以下基因:SOD1、C9orf72、ANG、FUS和TDP - 43。对27例ALS患者(22例SOD1阳性和5例SOD1阴性)和82例健康对照者进行了一系列广泛的经典神经心理学测试。241例ALS患者中共有37例(15.4%)携带检测基因的突变——17例家族性ALS患者中有16例(94.1%)呈阳性,224例明显散发性患者中有21例(9.4%)有致病突变。SOD1基因突变最为常见,占所有基因检测呈阳性的ALS患者的27例(73.0%)。SOD1阳性患者的主要临床特征为:下肢脊髓起病、常见括约肌和感觉障碍以及执行功能障碍综合征。在SOD1阳性患者中,我们注意到A145G患者发病稍早,L144F患者感觉和括约肌障碍占主导,而D90A患者有明显的感觉受累。SOD1阴性组由10例(27.0%)患者组成(6例C9orf72、2例ANG、1例TDP - 43以及1例携带三重FUS、C9orf72扩增和ANG变异的患者)。延髓受累和更广泛的神经心理学损害(包括执行、视觉空间和记忆困难)是SOD1阴性队列的主要特征。我们的结果表明,基于对患者的全面临床评估,可能对某些ALS基因型做出有意义的临床怀疑。

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Mutant SOD1 mediated pathogenesis of Amyotrophic Lateral Sclerosis.突变型超氧化物歧化酶1介导肌萎缩侧索硬化症的发病机制。
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