Department of Neurology, Fujian Medical University Union Hospital, Fuzhou, China; Institute of Clinical Neurology, Fujian Medical University, Fuzhou, China.
Department of Neurology, Fujian Provincial Hospital, Fuzhou, China.
Neurobiol Aging. 2021 Nov;107:168-173. doi: 10.1016/j.neurobiolaging.2021.05.017. Epub 2021 Jun 1.
Amyotrophic lateral sclerosis (ALS) is a progressive neurodegenerative disorder that predominately involves the motor neurons in the brain and spinal cord. The TARDBP gene, encoding TAR DNA-binding protein 43 (TDP-43) protein, has been identified as a major causative gene in ALS. In this study, we screened 275 SALS patients and 20 unrelated FALS probands for TARDBP mutations. We identified three TARDBP mutations in three SALS patients and two TARDBP mutations in two FALS probands, including a previously unreported mutation, p.K176I, in FALS patients consistent with frontotemporal dementia (FTD) and parkinsonism. The p.K176I mutation is the first mutation outside exon 6 of the TARDBP gene manifesting parkinsonism and the first TARDBP mutation manifesting parkinsonism identified in the Chinese population. Our results support that TARDBP mutations are one of the most common changes in both FALS and SALS in China. Patients with TARDBP mutations may have a broad phenotype spectrum of ALS, FTD, and parkinsonism. The TARDBP gene should be included in genetic screening for ALS with FTD, and/or parkinsonism.
肌萎缩侧索硬化症(ALS)是一种进行性神经退行性疾病,主要累及大脑和脊髓中的运动神经元。TAR DNA 结合蛋白 43(TDP-43)基因编码的 TARDBP 基因已被确定为 ALS 的主要致病基因。在这项研究中,我们对 275 名 SALS 患者和 20 名无关的 FALS 先证者进行了 TARDBP 基因突变筛查。我们在 3 名 SALS 患者和 2 名 FALS 先证者中发现了 3 种 TARDBP 突变,包括 FALS 患者中以前未报道的 p.K176I 突变,该突变与额颞叶痴呆(FTD)和帕金森病一致。p.K176I 突变是 TARDBP 基因外显子 6 以外第一个表现为帕金森病的突变,也是在中国人群中发现的第一个表现为帕金森病的 TARDBP 突变。我们的研究结果表明,TARDBP 突变是中国 FALS 和 SALS 中最常见的变化之一。TARDBP 突变患者可能具有 ALS、FTD 和帕金森病的广泛表型谱。TARDBP 基因应包含在具有 FTD 和/或帕金森病的 ALS 的遗传筛查中。