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快速眼动睡眠行为障碍患者一级亲属中伴有和不伴有孤立性快速动眼期无张力的神经退行性变的临床和神经影像学标志物:一项病例对照临床和多巴胺正电子发射断层扫描研究。

Clinical and neuroimaging markers of neurodegeneration in first-degree relatives of patients with REM sleep behavior disorder with and without isolated rapid eye movement sleep without atonia: A case-control clinical and dopamine PET study.

机构信息

Li Chiu Kong Family Sleep Assessment Unit, Department of Psychiatry, Faculty of Medicine, The Chinese University of Hong Kong, Shatin, Hong Kong, China.

Li Chiu Kong Family Sleep Assessment Unit, Department of Psychiatry, Faculty of Medicine, The Chinese University of Hong Kong, Shatin, Hong Kong, China.

出版信息

Parkinsonism Relat Disord. 2023 Feb;107:105271. doi: 10.1016/j.parkreldis.2022.105271. Epub 2022 Dec 26.

Abstract

OBJECTIVES

The current study aimed to examine the neurodegenerative implication of isolated REM sleep without atonia (RSWA) among first-degree relatives of patients with REM sleep behaviour disorder (RBD).

METHODS

This cross-sectional case-control study recruited three groups of subjects: First-degree relatives of RBD patients with isolated RSWA (n = 17), first-degree relatives of RBD patients without isolated RSWA (n = 18), and normal controls who did not have any RWSA and family history of RBD (n = 15). Prodromal Parkinson's Disease likelihood ratio by the updated MDS Research Criteria and striatal dopaminergic transmission function of the subjects as assessed by triple-tracer (18F-DOPA, 11C-Raclopride, and 18F-FDG) PET/CT scan were used as proxy markers of neurodegeneration.

RESULTS

In contrary to our hypothesis, the three groups did not differ in their pre- or post-striatal dopaminergic transmission function, and their Prodromal Parkinson's Disease likelihood ratio. However, they differed significantly in their frequency of a having first-degree relatives with Parkinson's disease or dementia of Lewy body (first-degree relativess with RSWA vs first degree relatives without RSWA vs normal controls = 58.8% vs 22.2% vs 0%, p = 0.001).

CONCLUSION

FDRs of RBD patients with isolated RSWA did not have increased neurodegenerative markers compared to FDRs of RBD patients without isolated RSWA and normal control, despite an paradoxical increase in frequency of Parkinson's disease or dementia of Lewy body among their family compared to FDRs of RBD patients without isolated RSWA. Further longitudinal follow-up study will be needed to ascertain their long-term prognosis.

摘要

目的

本研究旨在探讨 REM 睡眠期无动性发作(RSWA)的 REM 睡眠行为障碍(RBD)患者一级亲属的神经退行性变意义。

方法

本横断面病例对照研究招募了三组受试者:有孤立性 RSWA 的 RBD 患者一级亲属(n=17)、无孤立性 RSWA 的 RBD 患者一级亲属(n=18)和无 RSWA 且无 RBD 家族史的正常对照(n=15)。采用更新的 MDS 研究标准评估前驱期帕金森病可能性比和三重示踪剂(18F-DOPA、11C-racopride 和 18F-FDG)PET/CT 扫描评估受试者的纹状体多巴胺能传递功能,作为神经退行性变的替代标志物。

结果

与我们的假设相反,三组在纹状体前后多巴胺能传递功能及其前驱期帕金森病可能性比方面没有差异。然而,他们一级亲属中帕金森病或路易体痴呆的频率存在显著差异(有孤立性 RSWA 的一级亲属与无孤立性 RSWA 的一级亲属与正常对照组相比=58.8%比 22.2%比 0%,p=0.001)。

结论

与无孤立性 RSWA 的 RBD 患者一级亲属相比,有孤立性 RSWA 的 RBD 患者一级亲属的神经退行性标志物没有增加,尽管与无孤立性 RSWA 的 RBD 患者一级亲属相比,他们家族中帕金森病或路易体痴呆的频率呈反常增加。需要进一步的纵向随访研究来确定他们的长期预后。

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