King's College London, Institute of Pharmaceutical Science, 150 Stamford Street, London SE1 9NH, UK.
Swansea University, Singleton Park Campus, Swansea University Medical School, Swansea SA2 8PP, UK.
Hum Mol Genet. 2022 Aug 25;31(17):2918-2933. doi: 10.1093/hmg/ddac077.
Parkinson's disease is characterized by the deposition of α-synuclein, which leads to synaptic dysfunction, the loss of neuronal connections and ultimately progressive neurodegeneration. Despite extensive research into Parkinson's disease pathogenesis, the mechanisms underlying α-synuclein-mediated synaptopathy have remained elusive. Several lines of evidence suggest that altered nicotinamide adenine dinucleotide (NAD+) metabolism might be causally related to synucleinopathies, including Parkinson's disease. NAD+ metabolism is central to the maintenance of synaptic structure and function. Its synthesis is mediated by nicotinamide mononucleotide adenylyltransferases (NMNATs), but their role in Parkinson's disease is not known. Here we report significantly decreased levels of NMNAT3 protein in the caudate nucleus of patients who have died with Parkinson's disease, which inversely correlated with the amount of monomeric α-synuclein. The detected alterations were specific and significant as the expression levels of NMNAT1, NMNAT2 and sterile alpha and TIR motif containing 1 (SARM1) were not significantly different in Parkinson's disease patients compared to controls. To test the functional significance of these findings, we ectopically expressed wild-type α-synuclein in retinoic acid-differentiated dopaminergic SH-SY5Y cells that resulted in decreased levels of NMNAT3 protein plus a neurite pathology, which could be rescued by FK866, an inhibitor of nicotinamide phosphoribosyltransferase that acts as a key enzyme in the regulation of NAD+ synthesis. Our results establish, for the first time, NMNAT3 alterations in Parkinson's disease and demonstrate in human cells that this phenotype together with neurite pathology is causally related to α-synucleinopathy. These findings identify alterations in the NAD+ biosynthetic pathway as a pathogenic mechanism underlying α-synuclein-mediated synaptopathy.
帕金森病的特征是α-突触核蛋白的沉积,导致突触功能障碍、神经元连接丧失,最终导致进行性神经退行性变。尽管对帕金森病发病机制进行了广泛的研究,但α-突触核蛋白介导的突触病变的机制仍然难以捉摸。有几条证据表明,改变烟酰胺腺嘌呤二核苷酸(NAD+)代谢可能与包括帕金森病在内的突触核蛋白病有因果关系。NAD+代谢是维持突触结构和功能的核心。其合成由烟酰胺单核苷酸腺苷酰转移酶(NMNATs)介导,但它们在帕金森病中的作用尚不清楚。在这里,我们报告在患有帕金森病死亡的患者的尾状核中,NMNAT3 蛋白水平显著降低,这与单体α-突触核蛋白的量呈反比。这些改变是特异性和显著性的,因为与对照组相比,帕金森病患者的 NMNAT1、NMNAT2 和无菌α和 TIR 基序包含 1(SARM1)的表达水平没有显著差异。为了测试这些发现的功能意义,我们在外源性表达野生型α-突触核蛋白的维甲酸分化的多巴胺能 SH-SY5Y 细胞中检测到 NMNAT3 蛋白水平降低,加上神经突病理学,这可以通过 FK866 挽救,FK866 是烟酰胺磷酸核糖基转移酶的抑制剂,作为调节 NAD+合成的关键酶。我们的研究结果首次在帕金森病中确定了 NMNAT3 的改变,并在人类细胞中证明,这种表型加上神经突病理学与α-突触核蛋白病有因果关系。这些发现确定了 NAD+生物合成途径的改变是α-突触核蛋白介导的突触病变的致病机制。