Parkinson's Disease & Movement Disorders Unit, Neurology Service, Hospital Clínic I Universitari de Barcelona; IDIBAPS, CIBERNED (CB06/05/0018-ISCIII), ERN-RND, Institut Clínic de Neurociències (Maria de Maeztu Excellence Centre), Universitat de Barcelona. Barcelona, Catalonia, Spain; Lab of Parkinson Disease and Other Neurodegenerative Movement Disorders: Clinical and Experimental Research; Department of Neurology, Hospital Clínic de Barcelona, Institut D'Investigacions Biomèdiques August Pi I Sunyer (IDIBAPS), Institut de Neurociències, Universitat de Barcelona, Barcelona, Catalonia, Spain.
Parkinson's Disease & Movement Disorders Unit, Neurology Service, Hospital Clínic I Universitari de Barcelona; IDIBAPS, CIBERNED (CB06/05/0018-ISCIII), ERN-RND, Institut Clínic de Neurociències (Maria de Maeztu Excellence Centre), Universitat de Barcelona. Barcelona, Catalonia, Spain; Lab of Parkinson Disease and Other Neurodegenerative Movement Disorders: Clinical and Experimental Research; Department of Neurology, Hospital Clínic de Barcelona, Institut D'Investigacions Biomèdiques August Pi I Sunyer (IDIBAPS), Institut de Neurociències, Universitat de Barcelona, Barcelona, Catalonia, Spain.
Parkinsonism Relat Disord. 2022 Jun;99:33-41. doi: 10.1016/j.parkreldis.2022.05.006. Epub 2022 May 13.
Differential diagnosis between Parkinson's disease (PD) and atypical parkinsonisms (APs: multiple system atrophy[MSA], progressive supranuclear palsy[PSP], corticobasal degeneration[CBD]) remains challenging. Lately, cerebrospinal fluid (CSF) studies of neurofilament light-chain (NFL) and RT-QuIC of alpha-synuclein (α-SYN) have shown promise, but data on their combination with MRI measures is lacking.
(1) to assess the combined diagnostic ability of CSF RT-QuIC α-SYN, CSF NFL and midbrain/pons MRI planimetry in degenerative parkinsonisms; (2) to evaluate if biomarker-signatures relate to clinical diagnoses and whether or not unexpected findings can guide diagnostic revision.
We collected demographic and clinical data and set up α-SYN RT-QuIC at our lab in a cross-sectional cohort of 112 participants: 19 control subjects (CSs), 20PD, 37MSA, 23PSP, and 13CBD cases. We also determined CSF NFL by ELISA and, in 74 participants (10CSs, 9PD, 26MSA, 19PSP, 10CBD), automatized planimetric midbrain/pons areas from 3T-MRI.
Sensitivity of α-SYN RT-QuIC for PD was 75% increasing to 81% after revisiting clinical diagnoses with aid of biomarkers. Sensitivity for MSA was 12% but decreased to 9% with diagnostic revision. Specificities were 100% against CSs, and 89% against tauopathies raising to 91% with diagnostic revision. CSF NFL was significantly higher in APs. The combination of biomarkers yielded high diagnostic accuracy (PD vs. non-PD AUC = 0.983; MSA vs. non-MSA AUC = 0.933; tauopathies vs. non-tauopathies AUC = 0.924). Biomarkers-signatures fitted in most cases with clinical classification.
The combination of CSF NFL, CSF RT-QuIC α-SYN and midbrain/pons MRI measures showed high discriminant ability across all groups. Results opposite to expected can assist diagnostic reclassification.
帕金森病(PD)与非典型帕金森综合征(APs:多系统萎缩[MSA]、进行性核上性麻痹[PSP]、皮质基底节变性[CBD])的鉴别诊断仍然具有挑战性。最近,神经丝轻链(NFL)脑脊液(CSF)研究和α-突触核蛋白(α-SYN)的 RT-QuIC 显示出了希望,但关于它们与 MRI 测量相结合的数据尚缺乏。
(1)评估 CSF RT-QuIC α-SYN、CSF NFL 和中脑/脑桥 MRI 平面测量在退行性帕金森综合征中的联合诊断能力;(2)评估生物标志物特征是否与临床诊断相关,以及是否存在意外发现可以指导诊断修正。
我们在一个横断面队列中收集了 112 名参与者的人口统计学和临床数据,并在我们的实验室中建立了 α-SYN RT-QuIC:19 名对照组(CSs)、20 名 PD、37 名 MSA、23 名 PSP 和 13 名 CBD 病例。我们还通过 ELISA 确定了 CSF NFL,并在 74 名参与者(10 名 CSs、9 名 PD、26 名 MSA、19 名 PSP、10 名 CBD)中,从 3T-MRI 中自动测量中脑/脑桥区域。
α-SYN RT-QuIC 对 PD 的敏感性为 75%,在借助生物标志物重新审视临床诊断后,敏感性增加到 81%。MSA 的敏感性为 12%,但在诊断修正后降至 9%。特异性为 100%,与 CSs 相比,89%与 tauopathies 相比,诊断修正后为 91%。APs 中的 CSF NFL 显著升高。生物标志物的组合产生了较高的诊断准确性(PD 与非 PD AUC=0.983;MSA 与非 MSA AUC=0.933;tauopathies 与非 tauopathies AUC=0.924)。生物标志物特征与大多数临床分类相符。
CSF NFL、CSF RT-QuIC α-SYN 和中脑/脑桥 MRI 测量的组合在所有组别中均表现出了较高的鉴别能力。与预期相反的结果可以协助诊断重新分类。