Tagliafierro L, Chiba-Falek O
Department of Neurology, Duke University Medical Center, Durham, NC, 27710, USA.
Neurogenetics. 2016 Jul;17(3):145-57. doi: 10.1007/s10048-016-0478-0. Epub 2016 Mar 7.
Synucleinopathies are a group of neurodegenerative diseases that share a common pathological lesion of intracellular protein inclusions largely composed by aggregates of alpha-synuclein protein. Accumulating evidence, including genome wide association studies, has implicated alpha-synuclein (SNCA) gene in the etiology of synucleinopathies. However, the precise variants within SNCA gene that contribute to the sporadic forms of Parkinson's disease (PD), dementia with Lewy bodies (DLB), multiple system atrophy (MSA), and other synucleinopathies and their molecular mechanisms of action remain elusive. It has been suggested that SNCA expression levels are critical for the development of these diseases. Here, we review several model systems that have been developed to advance the understanding of the role of SNCA expression levels in the etiology of synucleinopathies. We also describe different molecular mechanisms that regulate SNCA gene expression and discuss possible strategies for SNCA down-regulation as means for therapeutic approaches. Finally, we highlight some examples that underscore the relationships between the genetic association findings and the regulatory mechanisms of SNCA expression, which suggest that genetic variability in SNCA locus is directly responsible, at least in part, to the changes in gene expression and explain the reported associations of SNCA with synucleinopathies. Future studies utilizing induced pluripotent stem cells (iPSCs)-derived neuronal lines and genome editing by CRISPR/Cas9, will allow us to validate, characterize, and manipulate the effects of particular cis-genetic variants on SNCA expression. Moreover, this model system will enable us to compare different neuronal and glial lineages involved in synucleinopathies representing an attractive strategy to elucidate-common and specific-SNCA-genetic variants, regulatory mechanisms, and vulnerable expression levels underlying synucleinopathy spectrum disorders. This forthcoming knowledge will support the development of precision medicine for synucleinopathies.
突触核蛋白病是一组神经退行性疾病,它们具有共同的病理病变,即细胞内蛋白质包涵体,主要由α-突触核蛋白聚集体组成。越来越多的证据,包括全基因组关联研究,表明α-突触核蛋白(SNCA)基因与突触核蛋白病的病因有关。然而,SNCA基因中导致散发性帕金森病(PD)、路易体痴呆(DLB)、多系统萎缩(MSA)和其他突触核蛋白病的确切变体及其分子作用机制仍不清楚。有人提出,SNCA表达水平对这些疾病的发展至关重要。在这里,我们综述了几种已开发的模型系统,以促进对SNCA表达水平在突触核蛋白病病因中作用的理解。我们还描述了调节SNCA基因表达的不同分子机制,并讨论了作为治疗方法下调SNCA的可能策略。最后,我们强调了一些例子,这些例子强调了基因关联研究结果与SNCA表达调控机制之间的关系,这表明SNCA基因座的遗传变异性至少部分直接导致了基因表达的变化,并解释了SNCA与突触核蛋白病的报道关联。利用诱导多能干细胞(iPSC)衍生的神经元系和CRISPR/Cas9进行基因组编辑的未来研究,将使我们能够验证、表征和操纵特定顺式遗传变体对SNCA表达的影响。此外,该模型系统将使我们能够比较参与突触核蛋白病的不同神经元和神经胶质谱系,这是一种有吸引力的策略,用于阐明突触核蛋白病谱系障碍潜在的共同和特定的SNCA遗传变体、调控机制和易损表达水平。这些即将获得的知识将支持突触核蛋白病精准医学的发展。