Department of Neurology, Faculty of Health Science, Medical University of Warsaw, Poland.
Department of Medical Genetics, Institute of Mother and Child, Warsaw, Poland.
J Clin Neurosci. 2020 Feb;72:501-503. doi: 10.1016/j.jocn.2019.12.018. Epub 2019 Dec 28.
Next Generation Sequencing (NGS), has now become a very powerful tool for decoding variants of genes involved in pathogenesis of number of human disorders. One of the challenges of this method is to decipher the real pathogenic variants from a number of identified, not related to the disorder in analyzed case. Another issue is recognition of new phenotypes previously unrecognized but related to new variants combinations' in known genes. The other aspect is the HGMD or ClinVar mutation databases usage in data interpretation. The aim of this paper is to discuss pathogenicity of p.(Glu121Lys) missense mutation in the TOR1A gene previously described as dystonia causing variant. The patient diagnosed with typical Parkinson disease and positive family history was included into analysis. Also the internal whole exome sequencing (WES) database containing 600 subjects who has performed WES due to different causes was searched. All subjects had WES performed on SureSelect Human All Exon v.6 enrichment, Illumina NovaSeq 6000 platform, (annotations according to internal Institute Mother and Child's pipeline). The TOR1A p.(Glu121Lys) heterozygous mutation was revealed in 1 patient diagnosed with PD and 2 healthy subjects who has no dystonia symptoms. To conclude the TOR1A p.Glu121Lys variant should not be recognized as clearly pathogenic now.
下一代测序 (NGS) 现已成为解码涉及许多人类疾病发病机制的基因变异的非常强大的工具。该方法的挑战之一是从大量已识别的与分析病例中疾病无关的变体中破译真正的致病性变体。另一个问题是识别以前未被识别但与已知基因中新型变体组合相关的新表型。另一个方面是在数据解释中使用 HGMD 或 ClinVar 突变数据库。本文旨在讨论 TOR1A 基因中先前描述为导致肌张力障碍的致病性 p.(Glu121Lys) 错义突变。将诊断为典型帕金森病且有阳性家族史的患者纳入分析。还搜索了包含 600 名因不同原因进行 WES 的内部全外显子组测序 (WES) 数据库。所有受试者均在 SureSelect Human All Exon v.6 富集、Illumina NovaSeq 6000 平台上进行 WES(根据内部研究所母子的管道进行注释)。在诊断为 PD 的 1 名患者和 2 名无肌张力障碍症状的健康受试者中发现了 TOR1A p.(Glu121Lys) 杂合突变。总之,TOR1A p.Glu121Lys 变体现在不应被视为明确的致病性。