A case-control-family study of idiopathic rapid eye movement sleep behavior disorder
Publication in refereed journal

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其它資訊
摘要Objective:
To determine the familial aggregation of idiopathic rapid eye movement sleep behavior disorder (iRBD), neurodegenerative diseases, and related biomarkers.

Methods:
A total of 404 and 387 first‐degree relatives of 102 patients with iRBD and of 89 controls were recruited, respectively. Among them, 204 and 208 relatives of patients and controls underwent face‐to‐face clinical assessment, whereas 97 and 75 relatives underwent further video‐polysomnographic assessment, respectively.

Results:
Compared with relatives of controls, relatives of patients demonstrated higher levels of RBD features, including chin tonic electromyography activity (mean = 1.5 ± 7.5 vs 0.3 ± 1.0, p = 0.04) and behavioral events (n [weighted %] = 12 [11.3] vs 2 [1.9], adjusted hazard ratio [aHR] = 7.69, 95% confidence interval [CI] = 1.54–33.33, p = 0.009) during rapid eye movement sleep, probable diagnosis (n [%] = 57 [14.9] vs 20 [4.9], aHR = 3.45, 95% CI = 1.96–6.25, p < 0.001), and definite diagnosis (n [weighted %] = 10 [8.4] vs 2 [1.4], aHR = 5.56, 95% CI = 1.16–25.00, p = 0.03). They also had higher risks of Parkinson disease (3.1% vs 0.5%, aHR = 5.88, 95% CI = 1.37–25.00, p = 0.02), dementia (6.9% vs 2.6%, aHR = 2.44, 95% CI = 1.15–5.26, p = 0.02), constipation (8.3% vs 2.4%, adjusted odds ratio = 4.21, 95% CI = 1.34–13.17, p = 0.01), and motor dysfunction (Movement Disorders Society Unified Parkinson's Disease Rating Scale part III motor score, mean = 1.9 ± 3.2 vs 0.9 ± 2.3, p = 0.002). The unaffected relatives of patients demonstrated a higher likelihood ratio of prodromal Parkinson disease (median [interquartile range] = 0.27 [1.19] vs 0.22 [0.51], p = 0.03).

Interpretation:
iRBD is familially aggregated from isolated features to full‐blown sleep disorder. Relatives of patients carry a higher risk of alpha‐synucleinopathy in terms of neurodegenerative diseases and prodromal markers, suggesting a familial aggregation and staging pathology of alpha‐synucleinopathy.
出版社接受日期11.02.2019
著者Liu Y, Zhang J, Lam SP, Zhou J, Yu MWM, Li SX, Chan JWY, Postuma RB, Mok VCT, Wing YK
期刊名稱Annals of Neurology
出版年份2019
月份4
卷號85
期次4
頁次582 - 592
國際標準期刊號0364-5134
語言美式英語

上次更新時間 2020-10-08 於 03:02