Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls

Gotthard G. Tribl, Mateus C. Trindade, Thais Bittencourt, Geraldo Lorenzi-Filho, Rosana Cardoso Alves, Daniel Ciampi de Andrade, Erich T. Fonoff, Edson Bor-Seng-Shu, Alexandre A. Machado, Carlos H. Schenck, Manoel J. Teixeira, Egberto R. Barbosa

Research output: Contribution to journalArticle

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Abstract

Objective: Quantitative data are reported on rapid eye movement (REM) sleep behavior disorder (RBD) in a cohort of predominantly neurological Wilson's disease (WD). Methods: A total of 41 patients with WD and 41 healthy, age- and gender-matched controls were studied by conducting face-to-face interviews, neurological and clinical examinations, laboratory tests, and WD- and RBD-specific scales. Video-polysomnography and quantification of REM sleep without atonia (RWA) were conducted in 35 patients and 41 controls. Results: Patients with WD showed significantly worse sleep quality, less sleep efficiency, increased wakefulness after sleep onset, and more arousals compared to healthy controls. Five patients with WD (four women) fulfilled the diagnostic criteria for RBD with significantly higher values in RWA, RBD Questionnaire-Hong Kong, and RBD Screening Questionnaire compared to patients with WD without RBD. In three patients with WD, RBD had manifested before any other symptom that could be attributed to WD. Percentage of RWA was significantly lower in WD without RBD than in WD with RBD, but still significantly increased compared to controls. Conclusions: RBD can be comorbid with WD. RWA is commonly present in WD, both in the presence or absence of clinical RBD. A causal connection is possible, though retrospective determination of RBD onset and the low number of patients do not allow a definitive conclusion at this point. However, screening for WD in idiopathic RBD is available at low cost and is recommended. Early-stage copper chelation therapy provides a highly effective treatment to prevent further WD manifestations and might also control the comorbid RBD.

Original languageEnglish (US)
Pages (from-to)179-185
Number of pages7
JournalSleep Medicine
Volume17
DOIs
StatePublished - Jun 10 2015

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REM Sleep Behavior Disorder
Hepatolenticular Degeneration
REM Sleep
Sleep
Chelation Therapy
Polysomnography
Wakefulness
Neurologic Examination
Hong Kong

Keywords

  • Movement disorders
  • Parasomnias
  • Parkinsonian syndromes
  • Rapid eye movement sleep behavior disorder
  • Sleep disorders
  • Wilson's disease

Cite this

Tribl, G. G., Trindade, M. C., Bittencourt, T., Lorenzi-Filho, G., Cardoso Alves, R., Ciampi de Andrade, D., ... Barbosa, E. R. (2015). Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls. Sleep Medicine, 17, 179-185. https://doi.org/10.1016/j.sleep.2015.09.003

Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls. / Tribl, Gotthard G.; Trindade, Mateus C.; Bittencourt, Thais; Lorenzi-Filho, Geraldo; Cardoso Alves, Rosana; Ciampi de Andrade, Daniel; Fonoff, Erich T.; Bor-Seng-Shu, Edson; Machado, Alexandre A.; Schenck, Carlos H.; Teixeira, Manoel J.; Barbosa, Egberto R.

In: Sleep Medicine, Vol. 17, 10.06.2015, p. 179-185.

Research output: Contribution to journalArticle

Tribl, GG, Trindade, MC, Bittencourt, T, Lorenzi-Filho, G, Cardoso Alves, R, Ciampi de Andrade, D, Fonoff, ET, Bor-Seng-Shu, E, Machado, AA, Schenck, CH, Teixeira, MJ & Barbosa, ER 2015, 'Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls', Sleep Medicine, vol. 17, pp. 179-185. https://doi.org/10.1016/j.sleep.2015.09.003
Tribl GG, Trindade MC, Bittencourt T, Lorenzi-Filho G, Cardoso Alves R, Ciampi de Andrade D et al. Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls. Sleep Medicine. 2015 Jun 10;17:179-185. https://doi.org/10.1016/j.sleep.2015.09.003
Tribl, Gotthard G. ; Trindade, Mateus C. ; Bittencourt, Thais ; Lorenzi-Filho, Geraldo ; Cardoso Alves, Rosana ; Ciampi de Andrade, Daniel ; Fonoff, Erich T. ; Bor-Seng-Shu, Edson ; Machado, Alexandre A. ; Schenck, Carlos H. ; Teixeira, Manoel J. ; Barbosa, Egberto R. / Wilson's disease with and without rapid eye movement sleep behavior disorder compared to healthy matched controls. In: Sleep Medicine. 2015 ; Vol. 17. pp. 179-185.
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abstract = "Objective: Quantitative data are reported on rapid eye movement (REM) sleep behavior disorder (RBD) in a cohort of predominantly neurological Wilson's disease (WD). Methods: A total of 41 patients with WD and 41 healthy, age- and gender-matched controls were studied by conducting face-to-face interviews, neurological and clinical examinations, laboratory tests, and WD- and RBD-specific scales. Video-polysomnography and quantification of REM sleep without atonia (RWA) were conducted in 35 patients and 41 controls. Results: Patients with WD showed significantly worse sleep quality, less sleep efficiency, increased wakefulness after sleep onset, and more arousals compared to healthy controls. Five patients with WD (four women) fulfilled the diagnostic criteria for RBD with significantly higher values in RWA, RBD Questionnaire-Hong Kong, and RBD Screening Questionnaire compared to patients with WD without RBD. In three patients with WD, RBD had manifested before any other symptom that could be attributed to WD. Percentage of RWA was significantly lower in WD without RBD than in WD with RBD, but still significantly increased compared to controls. Conclusions: RBD can be comorbid with WD. RWA is commonly present in WD, both in the presence or absence of clinical RBD. A causal connection is possible, though retrospective determination of RBD onset and the low number of patients do not allow a definitive conclusion at this point. However, screening for WD in idiopathic RBD is available at low cost and is recommended. Early-stage copper chelation therapy provides a highly effective treatment to prevent further WD manifestations and might also control the comorbid RBD.",
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AU - Tribl, Gotthard G.

AU - Trindade, Mateus C.

AU - Bittencourt, Thais

AU - Lorenzi-Filho, Geraldo

AU - Cardoso Alves, Rosana

AU - Ciampi de Andrade, Daniel

AU - Fonoff, Erich T.

AU - Bor-Seng-Shu, Edson

AU - Machado, Alexandre A.

AU - Schenck, Carlos H.

AU - Teixeira, Manoel J.

AU - Barbosa, Egberto R.

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N2 - Objective: Quantitative data are reported on rapid eye movement (REM) sleep behavior disorder (RBD) in a cohort of predominantly neurological Wilson's disease (WD). Methods: A total of 41 patients with WD and 41 healthy, age- and gender-matched controls were studied by conducting face-to-face interviews, neurological and clinical examinations, laboratory tests, and WD- and RBD-specific scales. Video-polysomnography and quantification of REM sleep without atonia (RWA) were conducted in 35 patients and 41 controls. Results: Patients with WD showed significantly worse sleep quality, less sleep efficiency, increased wakefulness after sleep onset, and more arousals compared to healthy controls. Five patients with WD (four women) fulfilled the diagnostic criteria for RBD with significantly higher values in RWA, RBD Questionnaire-Hong Kong, and RBD Screening Questionnaire compared to patients with WD without RBD. In three patients with WD, RBD had manifested before any other symptom that could be attributed to WD. Percentage of RWA was significantly lower in WD without RBD than in WD with RBD, but still significantly increased compared to controls. Conclusions: RBD can be comorbid with WD. RWA is commonly present in WD, both in the presence or absence of clinical RBD. A causal connection is possible, though retrospective determination of RBD onset and the low number of patients do not allow a definitive conclusion at this point. However, screening for WD in idiopathic RBD is available at low cost and is recommended. Early-stage copper chelation therapy provides a highly effective treatment to prevent further WD manifestations and might also control the comorbid RBD.

AB - Objective: Quantitative data are reported on rapid eye movement (REM) sleep behavior disorder (RBD) in a cohort of predominantly neurological Wilson's disease (WD). Methods: A total of 41 patients with WD and 41 healthy, age- and gender-matched controls were studied by conducting face-to-face interviews, neurological and clinical examinations, laboratory tests, and WD- and RBD-specific scales. Video-polysomnography and quantification of REM sleep without atonia (RWA) were conducted in 35 patients and 41 controls. Results: Patients with WD showed significantly worse sleep quality, less sleep efficiency, increased wakefulness after sleep onset, and more arousals compared to healthy controls. Five patients with WD (four women) fulfilled the diagnostic criteria for RBD with significantly higher values in RWA, RBD Questionnaire-Hong Kong, and RBD Screening Questionnaire compared to patients with WD without RBD. In three patients with WD, RBD had manifested before any other symptom that could be attributed to WD. Percentage of RWA was significantly lower in WD without RBD than in WD with RBD, but still significantly increased compared to controls. Conclusions: RBD can be comorbid with WD. RWA is commonly present in WD, both in the presence or absence of clinical RBD. A causal connection is possible, though retrospective determination of RBD onset and the low number of patients do not allow a definitive conclusion at this point. However, screening for WD in idiopathic RBD is available at low cost and is recommended. Early-stage copper chelation therapy provides a highly effective treatment to prevent further WD manifestations and might also control the comorbid RBD.

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KW - Parasomnias

KW - Parkinsonian syndromes

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