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Article: A case–control–family study of idiopathic rapid eye movement sleep behavior disorder

TitleA case–control–family study of idiopathic rapid eye movement sleep behavior disorder
Authors
Keywordsadult
case control study
clinical assessment
clinical feature
comparative study
Issue Date2019
PublisherJohn Wiley & Sons, Inc. The Journal's web site is located at http://www3.interscience.wiley.com/cgi-bin/jhome/76507645
Citation
Annals of Neurology, 2019, v. 85 n. 4, p. 582-592 How to Cite?
AbstractObjective: 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. Ann Neurol 2019;85:582-592.
Persistent Identifierhttp://hdl.handle.net/10722/294021
ISSN
2019 Impact Factor: 9.037
2015 SCImago Journal Rankings: 5.584
ISI Accession Number ID

 

DC FieldValueLanguage
dc.contributor.authorLiu, Y-
dc.contributor.authorZhang, J-
dc.contributor.authorLam, SP-
dc.contributor.authorZhou, J-
dc.contributor.authorYu, MWM-
dc.contributor.authorLi, SX-
dc.contributor.authorChan, JWY-
dc.contributor.authorPostuma, RB-
dc.contributor.authorMok, VCT-
dc.contributor.authorWing, YK-
dc.date.accessioned2020-11-23T08:25:14Z-
dc.date.available2020-11-23T08:25:14Z-
dc.date.issued2019-
dc.identifier.citationAnnals of Neurology, 2019, v. 85 n. 4, p. 582-592-
dc.identifier.issn0364-5134-
dc.identifier.urihttp://hdl.handle.net/10722/294021-
dc.description.abstractObjective: 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. Ann Neurol 2019;85:582-592.-
dc.languageeng-
dc.publisherJohn Wiley & Sons, Inc. The Journal's web site is located at http://www3.interscience.wiley.com/cgi-bin/jhome/76507645-
dc.relation.ispartofAnnals of Neurology-
dc.rightsPreprint This is the pre-peer reviewed version of the following article: [FULL CITE], which has been published in final form at [Link to final article using the DOI]. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions. Postprint This is the peer reviewed version of the following article: [FULL CITE], which has been published in final form at [Link to final article using the DOI]. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions.-
dc.subjectadult-
dc.subjectcase control study-
dc.subjectclinical assessment-
dc.subjectclinical feature-
dc.subjectcomparative study-
dc.titleA case–control–family study of idiopathic rapid eye movement sleep behavior disorder-
dc.typeArticle-
dc.identifier.emailLi, SX: shirleyx@hku.hk-
dc.identifier.authorityLi, SX=rp02114-
dc.description.naturelink_to_subscribed_fulltext-
dc.identifier.doi10.1002/ana.25435-
dc.identifier.pmid30761606-
dc.identifier.scopuseid_2-s2.0-85063075028-
dc.identifier.hkuros319102-
dc.identifier.volume85-
dc.identifier.issue4-
dc.identifier.spage582-
dc.identifier.epage592-
dc.identifier.isiWOS:000466415500014-
dc.publisher.placeUnited States-
dc.identifier.issnl0364-5134-

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