Patients with Primary Cervical Dystonia have Significant Sleep Impairment and Polysomnographic Abnormalities

Ray, Somdattaa; Kumar, Gagan; Kutty, Bindu M.; Pal, Pramod Kumar; Yadav, Ravi

doi:10.1007/s41782-021-00144-6

Cited by 4 publications

(10 citation statements)

References 29 publications

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“…lying down) in those with cranio-cervical dystonia which may be misclassified as a sleeping state [4]. Of these two studies, a total sleep time closely reflecting our results (470.5 min) was identified amongst a younger cohort (mean age: 42.8 years) [5], while in comparison to findings shown here reduced TST (368.6 min) were reported in a cohort closer matched in age (50.5 years) [4]. A recent study investigating the pathophysiological effects of BoNT treatment on sleep have also shown TST estimated using actigraphy was comparable to controls, although small sample size may explain observed differences (n = 6) [32].…”

Section: Discussionsupporting

confidence: 83%

“…These findings suggest that shared underlying mechanisms may give rise to the abnormal sleep architecture observed in this study and the motor features evident in AOIFCD. Interestingly, PSG-based studies have previously reported REM sleep change, including increased REM sleep latency and reduced REM sleep percentage in patients with cervical dystonia [4,5]; however, several oral medical therapies used in the management of the motor symptoms of dystonia (e.g. trihexyphenidyl and benzodiazepines) have been linked with decreased REM sleep duration [35,36], with these studies reporting 48% of the cohort prescribed trihexyphenidyl and 24% prescribed clonazepam [5].…”

Section: Discussionmentioning

confidence: 99%

“…BDI scores were not included in this model due to the low completion level; however, further assessments of mood would in key in any future studies. Of the few polysomnographic studies to date, PSG-studies have identified no differences in TST between cervical dystonia and control cohorts [ 4 , 5 , 32 ], although evidence suggests reduced muscle activity when in a relaxed state (i.e. lying down) in those with cranio-cervical dystonia which may be misclassified as a sleeping state [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

“…Associated non-motor symptoms are becoming increasingly recognised, with estimates between 33 to 70% of individuals experiencing sleep impairment with an associated reduction in quality of life [2,3]. To date, few studies have addressed sleep in AOIFCD using 'gold-standard' polysomnography (PSG), with those that have demonstrated reduced sleep efficiency, together with increased sleep latency, wake after sleep onset (WASO), REM sleep latency and percentage of time spent in rapid eye movement (REM) sleep, in comparison to controls [4][5][6]. However, PSG can be expensive and time consuming, with limitations in being able to extrapolate findings from single night recordings in an unfamiliar environment to the wider context of sleep.…”

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Use of remote monitoring and integrated platform for the evaluation of sleep quality in adult-onset idiopathic cervical dystonia

Bailey

Matthews²,

Szewczyk-krolikowski

et al. 2022

J Neurol

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Background Up to 70% of individuals diagnosed with adult-onset idiopathic focal cervical dystonia (AOIFCD) report difficulties with sleep. Larger cohort studies using wrist-worn accelerometer devices have emerged as an alternative to smaller polysomnography studies, in order to evaluate sleep architecture. Methods To measure activity during the sleep/wake cycle, individuals wore a consumer-grade wrist device (Garmin vivosmart 4) continuously over 7 days on their non-dominant wrist, while completing a daily sleep diary and standardised sleep and non-motor questionnaires via a dedicated app. Sleep measures were derived from the captured raw triaxial acceleration and heart rate values using previously published validated algorithms. Results Data were collected from 50 individuals diagnosed with AOIFCD and 47 age- and sex-matched controls. Those with AOIFCD self-reported significantly higher levels of excessive daytime sleepiness (p = 0.04) and impaired sleep quality (p = 0.03), while accelerometer measurements found the AOIFCD cohort to have significantly longer total sleep times (p = 0.004) and time spent in NREM sleep (p = 0.009), compared to controls. Overall, there was limited agreement between wearable-derived sleep parameters, and self-reported sleep diary and visual analogue scale records. Discussion This study shows the potential feasibility of using consumer-grade wearable devices in estimating sleep measures at scale in dystonia cohorts. Those diagnosed with AOIFCD were observed to have altered sleep architecture, notably longer total sleep time and NREM sleep, compared to controls. These findings suggest that previously reported disruptions to brainstem circuitry and serotonin neurotransmission may contribute to both motor and sleep pathophysiology.

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Section: Discussionsupporting

confidence: 83%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Use of remote monitoring and integrated platform for the evaluation of sleep quality in adult-onset idiopathic cervical dystonia

Bailey

Matthews²,

Szewczyk-krolikowski

et al. 2022

J Neurol

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“…Within the overall dystonia cohort, specific areas of sleep disturbance included insomnia, reduced sleep duration and daytime sleepiness, in part consistent with previous work [ 9 ]. However, the sleep questionnaires used during UKBB assessment differ from the standardised, sleep specific questionnaires typically used in previous studies of dystonia and lack the depth of understanding provided by investigations such as polysomnography [ 44 ].…”

Section: Discussionmentioning

confidence: 99%

Clinical and genotypic analysis in determining dystonia non-motor phenotypic heterogeneity: a UK Biobank study

et al. 2022

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The spectrum of non-motor symptoms in dystonia remains unclear. Using UK Biobank data, we analysed clinical phenotypic and genetic information in the largest dystonia cohort reported to date. Case–control comparison of dystonia and matched control cohort was undertaken to identify domains (psychiatric, pain, sleep and cognition) of increased symptom burden in dystonia. Whole exome data were used to determine the rate and likely pathogenicity of variants in Mendelian inherited dystonia causing genes and linked to clinical data. Within the dystonia cohort, phenotypic and genetic single-nucleotide polymorphism (SNP) data were combined in a mixed model analysis to derive genetically informed phenotypic axes. A total of 1572 individuals with dystonia were identified, including cervical dystonia (n = 775), blepharospasm (n = 131), tremor (n = 488) and dystonia, unspecified (n = 154) groups. Phenotypic patterns highlighted a predominance of psychiatric symptoms (anxiety and depression), excess pain and sleep disturbance. Cognitive impairment was limited to prospective memory and fluid intelligence. Whole exome sequencing identified 798 loss of function variants in dystonia-linked genes, 67 missense variants (MPC > 3) and 305 other forms of non-synonymous variants (including inframe deletion, inframe insertion, stop loss and start loss variants). A single loss of function variant (ANO3) was identified in the dystonia cohort. Combined SNP and clinical data identified multiple genetically informed phenotypic axes with predominance of psychiatric, pain and sleep non-motor domains. An excess of psychiatric, pain and sleep symptoms were evident across all forms of dystonia. Combination with genetic data highlights phenotypic subgroups consistent with the heterogeneity observed in clinical practice.

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Accelerometer‐derived sleep measures in idiopathic dystonia: A UK Biobank cohort study

Bailey,

Wadon,

Komarzynski

et al. 2023

Brain and Behavior

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BackgroundSleep disturbance is an increasingly recognized non‐motor trait in dystonia, with varying findings reported to date. Here, we examine sleep in a UK Biobank derived dystonia cohort using subjective self‐reported sleep symptoms and objective accelerometer‐derived sleep measures, with comparison to a control population.MethodsA total of 241 dystonia cases were compared to 964 matched controls in analysis of self‐reported sleep symptoms and changes in sleep architecture using wrist‐worn triaxial accelerometers.ResultsDystonia participants had poorer self‐reported sleep patterns compared to controls. Accelerometery measurements demonstrated later sleep times, reduced time in bed, and shifts in circadian rhythm. No association was observed with pain, and only limited relationships with psychiatric symptoms.DiscussionThis study demonstrates the utility of accelerometers in longer term evaluation of sleep in dystonia, for measurement of disturbance and response to treatment. Compared to controls, altered sleep and circadian rhythm were more common in dystonia patients which may contribute to the clinical phenotype.

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Patients with Primary Cervical Dystonia have Significant Sleep Impairment and Polysomnographic Abnormalities

Cited by 4 publications

References 29 publications

Use of remote monitoring and integrated platform for the evaluation of sleep quality in adult-onset idiopathic cervical dystonia

Use of remote monitoring and integrated platform for the evaluation of sleep quality in adult-onset idiopathic cervical dystonia

Clinical and genotypic analysis in determining dystonia non-motor phenotypic heterogeneity: a UK Biobank study

Accelerometer‐derived sleep measures in idiopathic dystonia: A UK Biobank cohort study

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