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Background: Restless legs syndrome (RLS) and periodic leg movements during sleep (PLMS) are prevalent sleep disorders with significant implications for health and well-being. While previous research has highlighted sex-related disparities in RLS and PLMS prevalence, comprehensive understanding of these differences across the lifespan remains limited. This study aims to explore sex differences in RLS and PLMS across diverse age groups, spanning ages 2 to over 80 years, and to investigate the underlying mechanisms influenced by sex hormones., Methods: A retrospective analysis was conducted on drug-free patients diagnosed with RLS, including 95 females (age range: 2-83.2 years) and 89 males (age range: 2-79.5 years). Polysomnographic recordings were analyzed to assess leg movement activity, including PLMS index and Periodicity index., Results: A more rapid increase in PLMS index was observed in women starting before age 10, plateauing lower than men until around age 55. An increase in women occurred after 55, lasting over a decade, while in men, PLMS index continued to rise after 75. Conversely, Periodicity index displayed a simpler pattern, increasing progressively from prepuberty to around 35 in males and 45-50 in females. Females maintained a slightly higher Periodicity index than males for over a decade after this age., Conclusion: These findings underscore the complex interplay between sex hormones, age, and sleep disorders, highlighting the need for tailored approaches to diagnosis and management across diverse demographic cohorts. Further research is warranted to elucidate the underlying mechanisms and develop targeted interventions to optimize sleep health outcomes., Competing Interests: Declaration of competing interest The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Raffaele Ferri reports financial support was provided by Italian Ministry of Health. If there are other authors, they declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Objectives: Poor sleep is frequently reported in children with neuromuscular diseases (NMD) and cerebral palsy (CP) however breathing disorders during sleep are often the clinical focus. Periodic limb movements (PLMs) have an increased prevalence in adults with NMD and may contribute to sleep disturbance in this population. We assessed the prevalence of PLMs in children with NMD or CP., Methods: Retrospective review of polysomnography (PSG) with leg electromyography in children age 1-18 years with NMD (including Duchenne muscular dystrophy, myotonic dystrophy, spinal muscular atrophy) or CP performed at a paediatric sleep centre 2004-2022., Results: Leg electromyography was available in at least 1 PSG in 239 children (125 NMD, 114 CP), and in 2 PSGs in 105 children (73 NMD, 32 CP). At initial PSG, 72 (30 %) were female with a median age 9y and respiratory disturbance index 3.5/h (interquartile range 1.3-9.9/h). Elevated PLM index (PLMI; >5/h) occurred in 9.6 % of each of the CP and NMD groups, quantified by initial PSG. Overall, PLMI increased from baseline (median 0, maximum 33/h) to follow-up (median 0, maximum 55.8/h; p < 0.05). In those with an elevated PLMI, arousal percentage attributable to PLMs was up to 25 % (median 7.5 %)., Conclusions: Elevated PLMI occurred at a higher prevalence in children with NMD and CP than reported in other clinic-referred paediatric populations. It is important that PLMs are not overlooked as identification and treatment may help improve sleep outcomes. Further research is required to understand the pathophysiology and consequences of PLMs specifically in this population., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Sleep is an integral and vital component of human life, contributing significantly to overall health and well-being, but a considerable number of people worldwide experience sleep disorders. Sleep disorder diagnosis heavily depends on accurately classifying sleep stages. Traditionally, this classification has been performed manually by trained sleep technologists that visually inspect polysomnography records. However, in order to mitigate the labor-intensive nature of this process, automated approaches have been developed. These automated methods aim to streamline and facilitate sleep stage classification. This study aims to classify sleep stages in a dataset comprising subjects with insomnia, PLM, and sleep apnea. The dataset consists of PSG recordings from the multi-ethnic study of atherosclerosis (MESA) cohort of the national sleep research resource (NSRR), including 2056 subjects. Among these subjects, 130 have insomnia, 39 suffer from PLM, 156 have sleep apnea, and the remaining 1731 are classified as good sleepers. This study proposes an automated computerized technique to classify sleep stages, developing a machine-learning model with explainable artificial intelligence (XAI) capabilities using wavelet-based Hjorth parameters. An optimal biorthogonal wavelet filter bank (BOWFB) has been employed to extract subbands (SBs) from 30 seconds of electroencephalogram (EEG) epochs. Three EEG channels, namely: Fz&#95;Cz, Cz&#95;Oz, and C4&#95;M1, are employed to yield an optimum outcome. The Hjorth parameters extracted from SBs were then fed to different machine learning algorithms. To gain an understanding of the model, in this study, we used SHAP (Shapley Additive explanations) method. For subjects suffering from the aforementioned diseases, the model utilized features derived from all channels and employed an ensembled bagged trees (EnBT) classifier. The highest accuracy of 86.8%, 87.3%, 85.0%, 84.5%, and 83.8% is obtained for the insomniac, PLM, apniac, good sleepers and complete datasets, respectively. Using these techniques and datasets, the study aims to enhance sleep stage classification accuracy and improve understanding of sleep disorders such as insomnia, PLM, and sleep apnea., Competing Interests: Declaration of Competing Interest None., (Copyright © 2024 IPEM. Published by Elsevier Ltd. All rights reserved.)

Purpose: Patients with early onset scoliosis are at high risk of sleep disordered breathing, sleep disruption, and adverse consequences of poor sleep. In this study, we aim to assess the prevalence of periodic limb movements of sleep in a cohort of children with early onset scoliosis and identify factors that correlate with the presence of periodic limb movements., Methods: This is a retrospective chart review of 40 patients with EOS (ages 1-17 years) who underwent a PSG from 2003 through 2019. Data collected included age, sex, and polysomnography parameters. Descriptive statistics were used: independent T test and Pearson correlation., Results: The average age was 9.6 years (SD 5.2); 22 were female. Eleven patients (27.5%) had elevated periodic limb movement index (PLMI) (≥ 5). Those with PLMI ≥ 5 had arousal index of 15.4 (SD 7.2) and those with normal PLMI having an arousal index of 9.4 (SD 4.9); this reached statistical significance (p < 0.05). Those with elevated PLMI spent 4.9 (SD 8.3) minutes with saturations below 88%, while those with normal PLMI spent 1.0 (SD 1.8); this was statistically significant. There was a moderate positive correlation between arousal index, hypoxemia, and PLMI., Conclusion: The study suggests that children with early onset scoliosis have higher frequency of periodic limb movements during sleep, and these may be correlated with increased arousal and with hypoxemia., (© 2024. The Author(s), under exclusive licence to Springer Nature Switzerland AG.)

Objective: Although manual scoring has been classically considered the gold standard to identify periodic leg movements (PLM), it is a very time consuming and expensive process, also subject to variability in interpretation. In the last decades, different authors have observed reasonably good agreement between automated PSG scoring algorithms and manual scoring in adults, according to established criteria. We aim to compare the automatic software analysis of our polysomnogram with the manual staging in children with sleep-disordered breathing., Methods: We performed a semiautomatic method, in which an experienced technician watched the video recording and removed from the automatic analysis those movements that did not correspond to true candidate leg movement (LM)., Results: A total of 131 PSGs were studied; applying the established criteria, 65 children were diagnosed of obstructive sleep apnea, and 66 presented snoring but with no sleep apnea. The mean age was 6.7 years (±1.7) and twenty-five children (19.08 %) had a PLMI >5/h. Statistical differences were found not only for PLMI (manual: 2.20 (0.7, 4.1) vs automatic (6.4 (3.85,9.5); p < 0.001), but for almost of all indexes assessed between the automatic and the manual scoring analysis. The level of concordance was only moderate for PLM index (0.63 [0.51-0.72]); showing that, unlike the articles published in the adult population, automatic analysis is not accurate in children and, manually or semi-automatically analysis as ours need to be done., Conclusion: It seems that PLM detection algorithm might work accurately but, the real need would be a true LM detection algorithm., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Excessive fragmentary myoclonus (EFM) is a frequent finding during routine video-polysomnography (VPSG). We aimed to automatically measure the prevalence of EFM according to current American Academy of Sleep Medicine (AASM) criteria, and the fragmentary myoclonus index (FMI) in sleep stage N1, N2, N3, rapid eye movement (REM) sleep and wake in a large patient population. A total of 500 VPSG recordings of patients admitted to the Sleep Laboratory, Department of Neurology, Medical University of Innsbruck, Austria, between May 1, 2022 and February 28, 2023, were included. EFM according to AASM criteria and FMI were computed by applying a previously validated algorithm. EFM was automatically detected in 121 of the 500 Sleep Laboratory patients (24.2%, 95% confidence interval 20.1%-28.9%). FMI increased with age, male gender, apnea-hypopnea-index (AHI), oxygen desaturation index (ODI), and periodic leg movements of sleep (PLMS) index. FMI was highest in REM sleep behaviour disorder (RBD), followed by neurodegenerative and internal medicine diseases, but the increase in the FMI was not explained by the disease itself but rather by the age and sex of the patients. Almost a quarter of our patient population had EFM. However, the prevalence of EFM does not allow the drawing of any conclusions about the pathophysiology of EFM or even the determination of a pathological FMI cut-off value. Associations of the FMI with age, sex, AHI, ODI and PLMS are in line with previous studies, but the FMI needs to be evaluated in different disease entities to learn more about its pathophysiology., (© 2023 The Authors. Journal of Sleep Research published by John Wiley & Sons Ltd on behalf of European Sleep Research Society.)

Sleep symptoms, including excessive sleepiness, are frequently reported by patients with functional motor disorders (FMD). We aimed to classify the comorbid sleep disorders in FMD, and to investigate the relationship between subjective sleepiness and objective measures of hypersomnia, comparing them with data from people with central hypersomnia. A total of 37 patients (mean [SD] age 46.4 [11.2] years) with clinically definite FMD, and 17 patients (mean [SD] age 41.1 [11.6] years) with central hypersomnia underwent structured medical and sleep history, neurological examination, polysomnography, multiple sleep latency test (MSLT), and questionnaires assessing sleepiness, fatigue, and depression. In all, 23 patients with FMD (62%) reported excessive daytime sleepiness. Evidence of specific sleep disorders was identified in our cohort, with 35% having restless legs syndrome; 49% obstructive sleep apnea; and 8% periodic limb movements in sleep; however, the presence of these disorders was not correlated with subjective sleepiness. Patients with FMD with self-reported sleepiness reported higher fatigue (p = 0.002), depression (p = 0.002), and had longer sleep latencies in the MSLT (p < 0.001) compared to the patients with central hypersomnia. No correlation was found between subjective and objective sleepiness in either group. Fatigue positively correlated with self-reported sleepiness in patients with FMD (p < 0.001). This study did not find objective correlates of increased sleepiness in patients with FMD. While sleep abnormalities were found to be common in FMD, they were not correlated with self-reports of excessive sleepiness. Positive correlations between self-reported sleepiness and fatigue support the current unified model of non-motor symptoms in FMD., (© 2023 The Authors. Journal of Sleep Research published by John Wiley & Sons Ltd on behalf of European Sleep Research Society.)

Introduction: The phenomenon known as periodic limb movements in sleep (PLMS) has been linked to a change in autonomic nervous system (ANS) activity and its effect on circulatory regulation. Autonomic dysfunction or dysregulation in patients with PLMS has been described in some domains; however, any relationship between heart rate variability (HRV) and PLMS has not been clearly established. HRV analysis is a recognised, non-invasive research method that describes the influence of the ANS on heart rate (HR). The aim of our study was to further investigate the dysregulation of autonomic HR control in patients with PLMS., Material and Methods: We undertook a retrospective analysis of the polysomnographic (PSG), demographic and medical data of five patients with a total number of 1,348 PLMS. We analysed HR, HRV HF, systolic blood pressure (SBP), and diastolic blood pressure (DBP) for 10 heartbeats before the series of PLMS and 10 consecutive heartbeats as beat-to-beat measurements. The presented method of using successive, short, 10 RR interval segments refers to the time-frequency measurement, which is very clear and useful for presenting changes in the calculated parameters over time and thereby illustrating their dynamics. This method allowed us to assess dynamic changes in HRV HF during successive PLMS series. Statistical analysis was performed using IBM SPSS Statistics (v. 28.0.0.0). The Kruskal-Wallis test was performed to find statistically significant changes from baseline., Results: No statistically significant changes in HR, SBP, or DBP were found in our group, although an increase in the value of the HRV HF was noted, suggesting an increase in intracardiac parasympathetic activity during the subsequent series of PLMS., Conclusions: Our study indicates an increase in parasympathetic activity during the appearance of successive PLMS, which, with the simultaneous lack of changes in HR, may suggest an increase in sympathetic activity, and therefore the appearance of so-called 'autonomic co-activation' resulting in the possibility of life-threatening cardiac events., Clinical Implications: Our findings add to the literature information regarding HRV in PLMS, and highlight the need for further studies to elucidate the effects of these conditions on the ANS, and on cardiovascular health.

Obstructive sleep apnoea (OSA) now affects one-seventh of the world's population. Treatment of even mild OSA can improve daytime sleepiness and quality of life. Recent modifications to uvulopalatopharyngoplasty may make it a more widely applicable treatment option in selected patients with OSA. Diet and exercise have effects on sleep apnoea severity independent of weight loss. Insomnia has become increasingly common during the coronavirus disease 2019 (COVID-19) pandemic., (© 2021 Asian Pacific Society of Respirology.)

Background: Restless legs syndrome (RLS) has been thought to increase the risk of hypertension, cardiovascular events, and all-cause mortality. Periodic limb movements in sleep (PLMS) can be observed in most patients with RLS. Using non-invasive physiologic measurement and analysis, including heart rate variability (HRV) analysis, we aimed to investigate sleep quality and sleep state stability., Method: A total of 53 healthy controls and 15 patients with RLS and PLMS were recruited. Patients with other sleep-related disorders such as obstructive sleep apnea (OSA) and major depressive disorder (MDD) were excluded. Each subject was evaluated using sleep and mood questionnaires and had to undergo polysomnography (PSG). HRV analysis was applied to assess autonomic function and analyze correlations with the severity of periodic leg movements (PLM). The power of different brainwaves was analyzed using electroencephalogram (EEG). Electromyogram (EMG) was also used to explore the temporal correlation between changes in HRV and leg movement events., Results: Compared with healthy controls, PLMS group had not only poorer perceived sleep and mood questionnaires scales but also reductions in parasympathetic-related HRV indices and increases in sympathetic-related HRV parameters. The changes were in proportion to the severity of PLM. Brainwaves and sleep stage which indicate "deep sleep" decreased in the PLMS group. There were no significant temporal correlations between changes in HRV and leg movement events., Conclusions: Our findings suggest that patients with RLS and PLMS have poorer subjective sleep and mood scales. Besides, objective sleep quality including HRV analysis and brainwaves analysis revealed reduced parasympathetic tone, increased sympathetic tone, and sleep disturbance, which reveal the possibility of a higher risk for secondary disease., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Objective: Subthalamic nucleus deep brain stimulation (STN DBS) has a positive effect on sleep quality, but its effect on wake functions is controversial. This study evaluated the longitudinal changes of the quality of sleep and excessive daytime sleepiness (EDS) in Parkinson's disease (PD) patients undergoing STN DBS and identify which factors are associated with the presence of EDS before and after STN DBS., Patient and Methods: A total of 33 PD patients who underwent bilateral STN DBS between July 2011 and October 2015 were recruited. We evaluated subjective sleep quality assessed by Parkinson's Disease Sleep Scale (PDSS) and EDS using Epworth Sleepiness Scale (ESS) preoperatively and 6 months, 1 year, and 3 years postoperatively., Results: There is a significant improvement in PDSS, and a noticeable change occurs immediately after the surgery. After DBS, the number of patients with persistent EDS gradually decreased, but patients with newly developed EDS were added. Baseline ESS score was highly correlated with EDS at 6 months and 1 year postoperatively, and older age of PD onset was highly associated with EDS at 1 year after DBS. At 3 years after DBS, the total PDSS score is a main contributing factor for EDS. There was no significant difference in dopamine agonist dose (agonist LED) and levodopa equivalent daily dose (LEDD) between groups with and without EDS at any time points., Conclusion: Bilateral STN DBS improves the subjective sleep quality, but EDS may improve or worsen. The risk factors for EDS change over time after STN DBS. Interestingly, dopaminergic medication did not affect EDS in DBS-treated PD patients., Competing Interests: Declaration of Competing Interest No conflicting relationship exists for the authors., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Motor activity during general anesthesia (GA) without neuromuscular blockade is often interpreted as reflecting insufficient anesthesia. Here we present the case of an octogenarian undergoing deep sclerectomy with opioid-sparing electroencephalography (EEG)-guided anesthesia. Periodic leg movements (PLM) appeared during ongoing surgery while the patient's raw EEG displayed a pattern of deep anesthesia, evidenced by burst suppression. Recognizing PLM in the context of opioid-sparing GA is of importance for anesthesiologists, as deep anesthesia is not necessarily associated with a decrease in motor activity.

Background: Evidence of the relationship between periodic limb movements during sleep (PLMS) and cerebral small vessel disease (cSVD) is limited and inconsistent. Here, we aimed to assess the independent association between PLMS and the different neuroimaging signatures of cSVD., Methods: Atahualpa residents aged more than or equal to 60 years enrolled in the Atahualpa Project undergoing polysomnography and MRI with time intervals less than or equal to 6 months were included. MRI readings focused on white matter hyperintensities (WMH) of presumed vascular origin, deep cerebral microbleeds (CMB), silent lacunar infarcts (LI), and more than 10 enlarged basal ganglia-perivascular spaces (BG-PVS). Data from single-night polysomnograms were interpreted according to recommendations of the American Academy of Sleep Medicine. Associations between the PLMS index and neuroimaging signatures of cSVD (as dependent variables) were assessed by means of logistic regression models, adjusted for relevant confounders., Results: A total of 146 individuals (mean age: 71.4 ± 7.5 years; 64% women) were included. A PLMS index more than or equal to 15 per hour were noted in 48 (33%) participants. Moderate-to-severe WMH were present in 33 individuals (23%), deep CMB in 9 (6%), silent LI in 16 (11%), and more than 10 BG-PVS in 44 (30%). In univariate analyses, silent LI (P = .035) and the presence of more than 10 enlarged BG-PVS (P = .034) were significantly higher among participants with a PLMS index more than or equal to 15 per hour. However, fully-adjusted multivariate models showed no significant association between PLMS index more than or equal to 15 per hour and any of the neuroimaging signatures of cSVD., Conclusions: This study shows no independent association between the PLMS index and neuroimaging signatures of cSVD in stroke-free community-dwelling older adults., (Copyright © 2019 Elsevier Inc. All rights reserved.)

Objective: Given the limited data available in the literature, the aim of this study was to examine the risk of resistant hypertension (RHT) associated with restless legs syndrome (RLS) and periodic limb movements during sleep (PLMS) in a large sample of treated hypertensive individuals., Methods: Demographic and polysomnographic (PSG) data from 673 treated hypertensive individuals recruited from the research database of the sleep laboratory of Erasme Hospital were analysed. After exclusion of the main causes of pseudo-resistance and secondary hypertension, RHT status was defined by the presence of an uncontrolled hypertension despite treatment with at least three antihypertensive agents (including a diuretic) from different classes in correct combination and at the highest tolerated doses or by the presence of controlled hypertension requiring the use of at least four antihypertensive agents. Logistic regression analyses were conducted to examine the risk of RHT associated with RLS and PLMS in treated hypertensive individuals., Results: After adjustment for major confounding factors associated with RHT, multivariate logistic regression analysis revealed that frequent RLS (≥2 episodes/week) combined with PLMS index ≥26/h [odds ratio (OR) 2.20; 95% confidence interval (CI) 1.35-3.61, p = 0.021] was a significant risk factor of RHT in treated hypertensive individuals., Conclusion: In treated hypertensive individuals, frequent RLS combined with PLMS index ≥26/h is associated with higher risk of RHT which suggests that this pathology may be a secondary cause of RHT (eg, obstructive sleep apnoea syndrome and insomnia with short sleep duration) justifying the establishment of effective treatments in this particular subpopulation., (Copyright © 2019 Elsevier B.V. All rights reserved.)

Multiple mechanisms may modulate an association between restless legs syndrome/Willis-Ekbom disease (RLS/WED) and cardiovascular disease (CVD), including chronic sleep deprivation, intermittent, periodic limb movements in sleep (PLMS)-related autonomic fluctuations and possible autonomic dysfunction intrinsically associated with RLS per se. The purpose of this paper is to review the existing RLS/WED literature focusing on the pathophysiologic evidence for possible associations between RLS/WED and PLMS with CVD and events (CVE). Specific intrinsic dysautonomic aspects of the disease, which may contribute to generating CVD, are separately discussed. The association between RLS/WED and both CV risk factors and CVD still remains elusive. Although several shared pathophysiological causes could explain these possible relationships, the emerging body of literature focusing on these disorders remains controversial. Not only longitudinal population-based studies and meta-analyses, but also more animal models and therapeutic interventions are needed in order to build a sufficiently robust body of evidence on this topic., (Copyright © 2019 Elsevier B.V. All rights reserved.)

None: Ventral thigh surface electromyography may be used to assess for periodic limb movements during sleep (PLMS) in a limb with an above-the-knee amputation. Presence of PLMS in the proximal portion of an amputated lower extremity supports theories of spinal and supraspinal mechanisms in PLMS generation, and demonstrates that intact distal motor efferent pathways and distal sensory afferent pathways are not absolutely necessary for the generation of periodic limb movements., Citation: Chada A, Hoque R. Periodic limb movements during sleep noted on ventral thigh surface electromyography in an above-the-knee amputated stump. J Clin Sleep Med. 2019;15(8):1183-1184., (© 2019 American Academy of Sleep Medicine.)

Objective: Fragmentary myoclonus (FM) is a polysomnographic motor phenomenon of unknown clinical relevance. This study investigates FM prevalence, gender differences, sleep stage distribution and association with clinical factors using recently introduced advanced FM scoring criteria., Methods: We analyzed polysomnographic recordings of 178 patients of a mixed sleep-disorder patient cohort. FM indices (FMI) of newly introduced 25 μV (FMI25) and standard 50 μV (FMI50) amplitude cut-offs were calculated., Results: FMI25 and FMI50 were higher in men compared to women. FMI were higher during wakefulness and lower during S3 compared to all other sleep stages, with stronger effects in men compared to women. FMI25 was correlated with higher age, lower mean oxygen saturation, lower sleep efficiency, higher periodic limb movement (PLM) index, shorter sleep period time and higher arousal index. Linear regression showed that age predicted higher FMI25 in both males and females. Additionally, higher arousal index predicted higher FMI25 in women only. FMI were not associated with the presence of sleep-related breathing disorders., Conclusions: We suggest FM represents a ubiquitous motor phenomenon occurring spontaneously during relaxed wakefulness and sleep, primarily in men and with advanced age., Significance: In women, particularly FMI25 may be a surrogate marker for more frequent arousals and sleep fragmentation., (Copyright © 2019 International Federation of Clinical Neurophysiology. Published by Elsevier B.V. All rights reserved.)

Study Objectives: Periodic limb movements (PLMs) have been associated with increased risk of stroke, but there is currently scarce research exploring this relationship in the setting of sickle cell disease (SCD). The aim of this study was to explore whether increased PLMs in children with SCD are associated with increased risk of cerebrovascular disease and to determine if there are any clinical or laboratory differences between children with SCD with elevated periodic limb movement index (PLMI) versus those with normal PLMI., Methods: This study is a comprehensive review of medical records of 129 children with SCD (aged ≤ 18 years) who had undergone polysomnography for evaluation of sleep-disordered breathing., Results: Elevated PLMI (PLMI > 5 events/h) was present in 42% (54/129) of children with SCD. Children with elevated PLMI were found to have higher percentage of hemoglobin S, lower total iron, higher arousal index and tendency toward elevated transcranial Doppler velocity ( P = .063, odds ratio = 3.9, 95% CI 0.93-16.22). While association between elevated PLMI and isolated cerebrovascular stenosis ( P = .050, odds ratio 5.6, 95% CI 1.0-31.10) trended toward significance, there was significantly greater proportion of children with elevated PLMI who had cerebrovascular stenosis with Moyamoya disease ( P = .046) as demonstrated by magnetic resonance imaging (MRI)., Conclusions: The prevalence of elevated PLMI in children with SCD was higher than in previously published data. Elevated PLMI was significantly associated with greater rates of cerebrovascular disease as detected by MRI., (Copyright © 2019 American Academy of Sleep Medicine. All rights reserved.)

Periodic limb movements (PLMs) may occur as a primary condition or be associated with other pathologies, such as sleep disorders. However, PLMs have not been described in comatose patients. We report the case of a 66-year-old man, with no history of sleep disorders, who presented PLMs during coma caused by an extensive right hemispheric abscess inducing midline shift. These movements were further characterized by video and electromyographic recordings, which displayed bilateral periodic bursts of the tibialis anterior muscles, occurring every 7 to 15 seconds, with no concomitant electroencephalographic correlate. After a long period of hospitalization, the patient eventually regained consciousness and PLMs seem to persist, only in sleep. To the best of our knowledge, this is the first report showing that PLMs may be observed in a setting where the networks supporting consciousness are lost, namely in a coma of structural etiology.

Study Objectives: Previous studies have shown that non-rapid eye movement (NREM) sleep parasomnias commonly coexist with restless legs syndrome (RLS) and periodic limb movement disorder (PLMD) in children, leading to speculation that RLS/PLMD may precipitate or worsen parasomnias. However, there are limited data about the effect of the treatment of RLS/PLMD on parasomnias in children. Hence, we performed this study to determine whether the treatment of RLS/PLMD with oral iron therapy is associated with improvement of parasomnias in children., Methods: A retrospective database was created for children with RLS/PLMD who were treated with iron therapy. These participants were followed for at least 1 year at Cincinnati Children's Hospital Medical Center. All participants had ferritin level testing and were treated with iron therapy. In addition, all participants underwent polysomnography before starting iron therapy for RLS/PLMD except for one participant who was already on iron but required a higher dose. Most participants underwent polysomnography after iron therapy., Results: A total of 226 participants were identified with the diagnosis of RLS/PLMD. Of these, 50 had parasomnias and 30 of them were treated with iron therapy. Of the 30 participants, RLS symptoms improved in 15 participants (50%) and resolution of parasomnias was noted in 12 participants (40%) participants after iron therapy. Repeat polysomnography after iron therapy was performed in 21 participants (70%). After iron therapy, there was a significant decrease in periodic limb movement index (17.2 ± 8.8 [before] versus 6.7 ± 7.3 [after] events/h, P < .001). In addition, there were significant decreases in PLMS (24.52 ± 9.42 [before] versus 7.50 ± 7.18 [after] events/h, P < .0001), PLMS-related arousals (4.71 ± 1.81 [before] versus 1.35 ± 1.43 [after] events/h, P < .0001), and total arousals (11.65 ± 5.49 [before] versus 8.94 ± 3.65 [after] events/h, P < .01) after iron therapy., Conclusions: Parasomnias are common in our cohort of children with RLS/PLMD. Iron therapy was associated with a significant improvement in periodic limb movement index, RLS symptoms, and resolution of a significant proportion of NREM sleep parasomnias, suggesting that RLS/PLMD may precipitate NREM sleep parasomnia., (© 2019 American Academy of Sleep Medicine.)

Background: Documentation of REM sleep without atonia is fundamental for REM sleep behavior disorder (RBD) diagnosis. The automated REM atonia index (RAI), Frandsen index (FRI) and Kempfner index (KEI) were proposed for this, but achieved moderate performances., New Method: Using sleep data from 27 healthy controls (C), 29 RBD patients and 36 patients with periodic limb movement disorder (PLMD), we developed and validated a new automated data-driven method for identifying movements in chin and tibialis electromyographic (EMG) signals. A probabilistic model of atonia from REM sleep of controls was defined and movements identified as EMG areas having low likelihood of being atonia. The percentages of movements and the median inter-movement distance during REM and non-REM (NREM) sleep were used for distinguishing C, RBD and PLMD by combining three optimized classifiers in a 5-fold cross-validation scheme., Results: The proposed method achieved average overall validation accuracies of 70.8% and 61.9% when REM and NREM, and only REM features were used, respectively. After removing apnea and arousal-related movements, they were 64.2% and 59.8%, respectively., Comparison With Existing Method(s): The proposed method outperformed RAI, FRI and KEI in identifying RBD patients and in particular achieved higher accuracy and specificity for classifying RBD., Conclusions: The results show that i) the proposed method has higher performances than the previous ones in distinguishing C, RBD and PLMD patients, ii) removal of apnea and arousal-related movements is not required, and iii) RBD patients can be better identified when both REM and NREM muscular activities are considered., (Copyright © 2018 Elsevier B.V. All rights reserved.)

Purpose: This study investigated the basal autonomic regulation in patients with obstructive sleep apnea (OSA) showing periodic limb movements in sleep (PLMS) emerging after therapy with continuous positive airway pressure (CPAP)., Methods: Data of patients with OSA undergoing a first polysomnography for diagnosis and a second polysomnography for therapy with CPAP were reviewed. Patients with OSA showing PLMS on the first polysomnography were excluded. By using heart rate variability analysis, epochs without any sleep events and continuous effects from the second polysomnography were retrospectively analyzed., Results: Of 125 eligible patients, 30 with PLMS after therapy with CPAP (PLMS group) and 30 not showing PLMS on both polysomnography (non-PLMS group) were randomly selected for the analysis. No significant differences in the demographic characteristics and variables of polysomnographies were identified between the groups. Although one trend of low root mean square of successive differences (RMSSD) between intervals of adjacent normal heart beats (NN intervals) in the PLMS group was observed, patients in the PLMS group had significantly low normalized high-frequency (n-HF) and high-frequency (HF) values, but high normalized low frequency (n-LF) and high ratio of LF to HF (LF/HF ratio). After adjustment for confounding variables, PLMS on the second polysomnography was significantly associated with RMSSD (β = - 6.7587, p = 0.0338), n-LF (β = 0.0907, p = 0.0148), n-HF (β = - 0.0895, p = 0.0163), log LF/HF ratio (β = 0.4923, p = 0.0090), and log HF (β = - 0.6134, p = 0.0199)., Conclusions: Patients with OSA showing PLMS emerging after therapy with CPAP may have a basal sympathetic predominance with potential negative cardiovascular effects.

Background: There is growing evidence of increased cardiovascular risk including hypertension in patients with periodic limb movements during sleep (PLMS). In a multiethnic cohort study, the association between prevalent hypertension and PLMS varied according to ethnicity. We evaluated whether PLMS are associated with hypertension in Koreans., Methods: We enrolled 1,163 subjects who had polysomnography (PSG) from 2 tertiary hospitals. All subjects completed a sleep questionnaire before the PSG study. Coincidental hypertension was recorded according to past medical history. We analyzed the association between periodic limb movement index (PLMI), periodic limb movement associated with arousal index (PLMAI), and coincidental hypertension. Covariates were age, sex, body mass index (BMI), restless legs syndrome, apnea-hypopnea index (AHI), arousal index, and average oxygen saturation., Results: A total of 304 subjects (26.1%) had hypertension. The proportion of subjects with hypertension in the PLMI ≥ 15 category was higher than that in the PLMI < 15 category (32.4% vs. 25.0%; P = 0.04). The proportion of subjects with hypertension in the PLMAI ≥ 1 category was 32.6%, which was higher than that in the PLMAI < 1 category (24.6%; P = 0.02). In a multivariate regression model, neither PLMI (odds ratio [OR], 1.12; 95% confidence interval [CI] 0.75-1.68) nor PLMAI (OR, 1.21; 95% CI 0.83-1.76) were associated with hypertension. Statistical significance was found between coincidental hypertension and the following variables: age, smoking history, BMI, and AHI., Conclusions: In a retrospective hospital-based study, there was no association between coincidental hypertension and PLMI/PLMAI in Koreans.

Rapid eye movement (REM) sleep without atonia detection is a prerequisite for diagnosis of REM sleep behavior disorder (RBD). As the visual gold standard method is time-consuming and subjective, several automated methods have been proposed. This study aims to compare their performances: The REM atonia index (RAI), the supra-threshold-REM-activity metric, the Frandsen index, the short/long muscle activity indices, and the Kempfner index algorithms were applied to 27 healthy control participants (C), 25 patients with Parkinson's disease (PD) without RBD (PD-RBD), 29 patients with PD and RBD (PD + RBD), 29 idiopathic patients with RBD, and 36 patients with periodic limb movement disorder (PLMD). The indices were calculated in various configurations: (1) considering all muscle activities; (2) excluding the ones related to arousals; (3) excluding the ones during apnea events; (4) excluding the ones before and after apnea events; (5) combining configurations 2 and 3; and (6) combining configurations 2 and 4. For each of these configurations, the discrimination capability of the indices was tested for the following comparisons: (1) (C, PD-RBD, PLMD) vs (PD + RBD, RBD); (2) C vs RBD; (3) PLMD vs RBD; (4) C vs PD-RBD; (5) C vs PLMD; (6) PD-RBD vs PD + RBD; and (7) C vs PLMD vs RBD. Results showed varying methods' performances across the different configurations and comparisons, making it impossible to identify the optimal method and suggesting the need of further improvements. Nevertheless, RAI seems the most sensible one for RBD detection. Moreover, apnea and arousal-related movements seem not to influence the algorithms' performances in patients' classification.

Aims: Nocturia, or waking up at night to void, is a highly prevalent and bothersome lower urinary tract symptom. However, the applied treatment modalities do not improve symptoms in about half of the patients. The aim of this report is to generate new ideas for future nocturia research, with special emphasis on the role of sleep physiology and sleep disorders., Methods: The following is a report of the presentations and subsequent discussion of the Nocturia Think Tank session at the annual meeting of the International Consultation on Incontinence Research Society (ICI-RS), which took place in September 2015 in Bristol. General information about the organization of the ICI-RS meeting can be found on the website "www.ici-rs.org." An overview of challenges within the existing evidence, future research ideas, and results of research with regard to nocturia and sleep were presented., Results and Conclusion: In order to optimize the management of nocturia and nocturnal polyuria (NP), future research has to focus on the development of unambiguous terminology regarding nocturia and NP, the role of renal function profiles and simplified frequency volume charts as guidance of individualized therapy and the role of sleep disorders such as periodic limb movements during sleep and habitual voiding as a response to awakening. Neurourol. Urodynam. 37:2048-2052, 2018. © 2016 Wiley Periodicals, Inc., (© 2016 Wiley Periodicals, Inc.)

Parasomnias and sleep-related movement disorders are important problems in older adults. Sleep paralysis is rare, but may occur in families. In a minority of patients with disorders of arousal, the episodes persist until the age of 70. Zolpidem and other medications may induce sleepwalking and sleep-related eating. Most patients with idiopathic rapid eye movement (REM) sleep behavior disorder eventually develop Parkinson's disease or dementia with Lewy bodies. Anti-IgLON5 disease includes abnormal behaviors in NREM and REM sleep. Restless legs syndrome prevalence increases with age. A severe form of periodic limb movements in sleep may mimic REM sleep behavior disorder., (Copyright © 2017 Elsevier Inc. All rights reserved.)

Study Objectives: To define statistically the upper limit of the intermovement interval (IMI, the time interval between the onset of consecutive movements) of periodic leg movements during sleep (PLMS)., Methods: We computed the IMI distribution of a large sample (n = 141) of patients with restless legs syndrome (RLS) and analyzed it with two independent approaches, based on fitting either empirical functions or normal and exponential functions to the data., Results: The two fitting approaches consistently pointed to an upper limit of the PLMS IMI in the range between 50 and 60 s. Decreasing the upper limit of PLMS IMI from 90 to 60 s evidently decreased the PLMS index in patients with RLS and control participants; nevertheless, the PLMS index remained significantly higher in RLS vs. control participants. Shifting the upper limit of PLMS IMI to 60 s did not significantly modify the effectiveness of discrimination of PLMS between controls and patients with RLS., Conclusion: These results seem to indicate that a conservative, yet data-driven upper limit for IMI contributing to the PLMS in patients with RLS might be 60 s instead of 90 s, as recommended by the present guidelines.

Objective: Periodic limb movements during sleep (PLMS) are prevalent in the general population, but their impact on sleep and association with cardiometabolic disorders are a matter of debate., Methods: Data from 2162 participants (51.2% women, mean age 58.4 ± 11.1 years) of the population-based HypnoLaus study (Lausanne, Switzerland) were collected. Subjective sleep complaints and habits were assessed using the Pittsburgh Sleep Quality Index and the Epworth Sleepiness Scale (ESS). Participants underwent a full polysomnography (PSG) at home and were evaluated for the presence of hypertension, diabetes, and metabolic syndrome., Results: Participants with a PLMS index (PLMSI) > 15/h (28.6% of the sample) had longer subjective sleep latency (18.6 ± 17.2 vs. 16.1 ± 14.3 min, p = 0.014) and duration (7.1 ± 1.2 vs. 6.9 ± 1.1 h, p < 0.001) than participants with PLMSI ≤ 15/h. At the PSG, they spent more time in stage N2 sleep (49.0 ± 11.2 vs. 45.5 ± 9.8%, p < 0.001), less in stage N3 (17.6 ± 8.2 vs. 20.6 ± 8.4%, p < 0.001) and in REM sleep (20.3 ± 6.4 vs. 22.4 ± 6.0%, p < 0.001), and exhibited longer REM latency (104.2 ± 70.2 vs. 91.7 ± 58.6 min, p < 0.001) and higher arousal index (26.5 ± 12.3 vs. 19.2 ± 9.7 n/h, p < 0.001). Participants with a PLMSI > 15/h had a lower ESS scores and higher prevalence of hypertension, diabetes, and metabolic syndrome. Multivariate analysis adjusting for confounding factors confirmed the independent association of PLMSI > 15/h with subjective sleep latency and duration, and with objective sleep structure disturbances. However, the associations with sleepiness and cardiovascular risk factors disappeared., Conclusions: In our large middle-age European population-based sample, PLMSI > 15/h was associated with subjective and objective sleep disturbances but not with sleepiness, hypertension, diabetes, or metabolic syndrome., (Copyright © 2017 Elsevier B.V. All rights reserved.)

Periodic limb movements (PLMs) during sleep increase with age and are associated with striatal neurodegeneration and dopamine deficiency. Limb movements are often associated with disruptions to non-rapid eye movement (NREM) sleep. Motor skill memory consolidation recruits the striatum, and learning-dependent striatal activation is associated with NREM sleep. Therefore, we investigated whether de novo individuals who significantly experience elevated levels of PLMs but have not been formally diagnosed with periodic limb movement disorder had learning and sleep-related memory deficits and whether these deficits were related to sleep quality and symptom severity. In total, 14 adults with significantly elevated PLMs (PLM condition), 15 age-matched controls (CTRL), and 14 age-matched "disturbed" sleep (through induced leg movements) controls (CTRL-ES) participated. The participants were trained (PM) and retested (AM) on procedural motor sequence learning (MSL) and declarative paired associates memory tasks. Baseline sleep quality was significantly worse in PLM than in CTRL. Despite the continued presence of PLMs in the PLM condition on the experimental night, remarkably, sleep quality improved and arousals decreased, vs. baseline, and did not differ from CTRL. MSL was significantly slower in the PLM condition than in CTRL at training but surprisingly exhibited overnight performance gains, which correlated with reduced arousals. As predicted, CTRL but not CTRL-ES had overnight gains in MSL. Taken together, this suggests that in the PLM condition, sleep quality was normalized following MSL, where they derived the same benefit of sleep to procedural memory consolidation as in CTRL. Sleep did not benefit declarative memory. Although preliminary, these results suggest that MSL in individuals with PLMs may provide a benefit to sleep, which in turn may benefit memory consolidation., (Copyright © 2017 Elsevier B.V. All rights reserved.)

Background: Growing evidence indicates that periodic limb movements of sleep (PLMS) may be related to increased risk of developing cardiovascular disease. However, the association of PLMS with atrial fibrillation (AF) is unclear, especially in patients with sleep-disordered breathing (SDB). This study sought to investigate whether PLMS were associated with increased AF prevalence, independent of established risk factors., Methods: We performed a cross-sectional study of patients who underwent attended polysomnography at Mayo Clinic from 2011 to 2014. The association of PLMS with AF prevalence was estimated by using logistic regression models., Results: 15,414 patients were studied, 76.3% of individuals with SDB defined by apnea-hypopnea index (AHI) ≥5/h, and 15.3% with a diagnosis of AF. In univariate logistic modelling, individuals with periodic limb movement index (PLMI) ≥30/h had higher odds of AF (odds ratio [OR] 1.96, 95% confidence interval [CI]1.79-2.16, p<0.001) when compared to patients with PLMI <15/h. After multivariate adjustment (for age, race, sex, history of smoking, hypertension, diabetes, coronary artery disease, heart failure, cerebrovascular disease, renal disease, iron deficiency anemia, chronic obstructive pulmonary disease, AHI, arousal index), in mild SDB patients, a PLMI ≥30/h or periodic limb movement arousal index (PLMAI) ≥5/h had significantly higher odds of AF than those with PLMI <15/h (OR 1.21, 95% CI 1.00-1.47, p=0.048) or PLMAI <1/h (OR 1.27, 95% CI 1.03-1.56, p=0.024)., Conclusions: Frequent PLMS are independently associated with AF prevalence in patients with mild SDB. Further studies are needed to better understand the relationship with incident AF., (Copyright © 2017 Elsevier B.V. All rights reserved.)

Restless legs syndrome (RLS) is a relatively common neurological disorder in childhood, although it is usually overlooked due to the atypical presentation in children and associated comorbid conditions that may affect its clinical presentation. Here, we aimed to perform, for the first time, a systematic review of studies reporting the association between RLS in children and adolescents (<18 y) and somatic or neuropsychiatric conditions. We searched for peer-reviewed studies in PubMed, Ovid (including PsycINFO, Ovid MEDLINE ® , and Embase), Web of Knowledge (Web of Science, Biological abstracts, BIOSIS, FSTA) through November 2015, with no language restrictions. We found 42 pertinent studies. Based on the retrieved studies, we discuss the association between RLS and a number of conditions, including growing pains, kidney disease, migraine, diabetes, epilepsy, rheumatologic disorders, cardiovascular disease, liver and gastrointestinal disorders, and neuropsychiatric disorders (e.g., attention deficit hyperactivity disorder (ADHD), depression, and conduct disorder). Our systematic review provides empirical evidence supporting the notion that RLS in children is comorbid with a number of somatic and neuropsychiatric conditions. We posit that the awareness on comorbid diseases/disorders is pivotal to improve the diagnosis and management of RLS and might suggest fruitful avenues to elucidate the pathophysiology of RLS in children., (Copyright © 2016 Elsevier Ltd. All rights reserved.)

Purpose of Review: Review of the literature pertaining to clinical presentation, classification, epidemiology, pathophysiology, diagnosis, and treatment of sleep-related movement disorders and disturbances of motor control., Recent Findings: Sleep-related movement disorders and disturbances of motor control are typically characterized by positive motor symptoms and are often associated with sleep disturbances and consequent daytime symptoms (e.g. fatigue, sleepiness). They often represent the first or main manifestation of underlying disorders of the central nervous system, which require specific work-up and treatment. Diverse and often combined cause factors have been identified. Although recent data provide some evidence regarding abnormal activation and/or disinhibition of motor circuits during sleep, for the majority of these disorders the pathogenetic mechanisms remain speculative. The differential diagnosis is sometimes difficult and misdiagnoses are not infrequent. The diagnosis is based on clinical and video-polysomnographic findings. Treatment of sleep-related motor disturbances with few exceptions (e.g. restless legs/limbs syndrome) are based mainly on anecdotal reports or small series., Summary: More state-of-the-art studies on the cause, pathophysiology, and treatment of sleep-related movement disorders and disturbances of motor control are needed.

Restless legs syndrome is a frequent neurological disorder with potentially serious and highly distressing treatment complications. The role and potential implications of periodic leg movements during sleep range from being a genetic risk marker for restless legs syndrome to being a cardiovascular risk factor. The diagnosis of restless legs syndrome in patients with daytime movement disorders is challenging and restless legs syndrome needs to be differentiated from other sleep-related movement disorders. This article provides an update on the diagnosis of restless legs syndrome as an independent disorder and the role of periodic leg movements and reviews the association of restless legs syndrome with Parkinson's disease and other movement disorders. © 2017 International Parkinson and Movement Disorder Society., (© 2017 International Parkinson and Movement Disorder Society.)

Purpose: Periodic limb movements in sleep (PLMS) are related to arousal, sympathetic activation, and increases in blood pressure (BP), but whether they are part of the arousal process or causative of it is unclear. Our objective was to assess the temporal distribution of arousal-related measures around PLMS., Methods: Polysomnographic recordings of six patients with restless legs syndrome were analyzed. We analyzed 15 PLMS, plus three 5-s epochs before and after each movement, for every patient. Mean values per epoch of blood pressure (BP), heart rate (HR), and electroencephalographic (EEG) power were calculated. For each patient, six 5-s epochs of undisturbed sleep were analyzed as controls., Results: Alpha + beta EEG power, systolic BP, and HR were significantly increased following PLMS. The EEG power and HR increases were noticed in the first epoch after PLMS, whereas that of systolic BP was observed in the second and third epochs following a PLMS. No significant changes occurred in the epochs of undisturbed sleep., Conclusions: The results suggest that PLMS are followed by arousal-related nervous system events. Given the high frequency of PLMS throughout the night, they could be a potential risk factor for nocturnal arrhythmias and hypertension, in addition to causing sleep deprivation.

Study Objectives: To describe a group of patients referred because of abnormal sleep behaviors that were suggestive of rapid eye movement (REM) sleep behavior disorder (RBD) in whom video-polysomnography ruled out RBD and showed the reported behaviors associated with vigorous periodic limb movements during sleep (PLMS)., Aims and Methods: Clinical history and video-polysomnography review of patients identified during routine visits in a sleep center., Results: Patients were 15 men and 2 women with a median age of 66 (range: 48-77) years. Reported sleep behaviors were kicking (n = 17), punching (n = 16), gesticulating (n = 8), falling out of bed (n = 5), assaulting the bed partner (n = 2), talking (n = 15), and shouting (n = 10). Behaviors resulted in injuries in 3 bed partners and 1 patient. Twelve (70.6%) patients were not aware of displaying abnormal sleep behaviors that were only noticed by their bed partners. Ten (58.8%) patients recalled unpleasant dreams such as being attacked or chased. Video-polysomnography showed (1) frequent and vigorous stereotyped PLMS involving the lower limbs, upper limbs, and trunk (median PLMS index 61.2; median PLMS index in NREM sleep 61.9; during REM sleep only 8 patients had PLMS and their median PLMS index in REM sleep was 39.5); (2) abnormal behaviors (e.g., punching, groaning) during some of the arousals that immediately followed PLMS in NREM sleep; and (3) ruled out RBD and other sleep disorders such as obstructive sleep apnea. Dopaminergic agents were prescribed in 14 out of the 17 patients and resulted in improvement of abnormal sleep behaviors and unpleasant dreams in all of them. After dopaminergic treatment, follow-up video-polysomnography in 7 patients showed a decrease in the median PLMS index from baseline (108.9 vs. 19.2, p = .002) and absence of abnormal behaviors during the arousals., Conclusions: Abnormal sleep behaviors and unpleasant dreams simulating RBD symptomatology may occur in patients with severe PLMS. In these cases, video-polysomnography ruled out RBD and identified prominent PLMS followed by arousals containing abnormal behaviors. Our cases represent an objectively documented subtype of periodic limb movement disorder causing abnormal sleep behaviors., (© Sleep Research Society 2016. Published by Oxford University Press on behalf of the Sleep Research Society. All rights reserved. For permissions, please e-mail journals.permissions@oup.com.)

Periodic leg movements during sleep (PLMS) are the most important objective finding in restless legs syndrome (RLS). During the last decade, PLMS have been very important for the assessment and comprehension of their pathophysiological correlates, which have been paralleled by the emergence of new computer-assisted and data-driven rules for their identification, scoring, and analysis. The present article focused on the most relevant PLMS-related findings of the last decade, and sought to provide a coherent and comprehensive overview on this enigmatic motor phenomenon. First, a clear description was made on the identification, quantification, and scoring of PLMS and their associated events. This was followed by a description of the current knowledge of their neurophysiologic aspects. Then, the typical phenotype of genuine PLMS in RLS and other clinical conditions was described, allowing for their careful separation from other sleep leg motor activities. In addition, the most recent findings on the genetics of PLMS were briefly summarized, followed by the current evidence on their clinical correlates, which is another rapidly advancing field of research. The description of the specific aspects of PLMS in children was also carefully reported, with important clues on their evaluation in this age group. Finally, further research was proposed, which may lead to consideration of PLMS as a clinically significant concern, independent of the association with RLS., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Study Objectives: Emerging evidence suggests that periodic limb movements (PLMs) may contribute to the development of cerebrovascular disease. White matter hyperintensities (WMHs), a widely accepted biomarker for cerebral small vessel disease, are associated with incident stroke and death. We evaluated the association between increased PLM indices and WMH burden in patients presenting with stroke or transient ischemic attack (TIA), while controlling for vascular risk factors and stroke severity., Methods: Thirty patients presenting within 2 weeks of a first-ever minor stroke or high-risk TIA were prospectively recruited. PLM severity was measured with polysomnography. WMH burden was quantified using the Age Related White Matter Changes (ARWMC) scale based on neuroimaging. Partial Spearman's rank-order correlations and multiple linear regression models tested the association between WMH burden and PLM severity., Results: Greater WMH burden was correlated with elevated PLM index and stroke volume. Partial Spearman's rank-order correlations demonstrated that the relationship between WMH burden and PLM index persisted despite controlling for vascular risk factors. Multivariate linear regression models revealed that PLM index was a significant predictor of an elevated ARWMC score while controlling for age, stroke volume, stroke severity, hypertension, and apnea-hypopnea index., Conclusion: The quantity of PLMs was associated with WMH burden in patients with first-ever minor stroke or TIA. PLMs may be a risk factor for or marker of WMH burden, even after considering vascular risk factors and stroke severity. These results invite further investigation of PLMs as a potentially useful target to reduce WMH and stroke burden., (© Sleep Research Society (SRS) 2016. All rights reserved. For permissions, please email: journals.permissions@oup.com)

Objectives: Primary restless leg syndrome (RLS) and periodic limb movements in sleep (PLMS) frequently co-exist, obscuring the boundaries between the two conditions. In such instances, a study of secondary cases with focal lesions such as post-stroke RLS and PLMS (psRLS and psPLMS, respectively) can be helpful in identifying characteristics of the individual conditions., Materials and Methods: Patients who had suffered strokes and who subsequently developed psRLS or psPLMS were recruited. To determine the overall features of psRLS/PLMS, historical cases were selected from the literature. All cases with either psRLS or psPLMS alone were further analyzed to elucidate the distinctive pathomechanisms of the two conditions., Results: Six patients with either psRLS or psPLMS were recruited from our hospital; two patients had both conditions contemporaneously. The literature contains details on 30 cases of psRLS or psPLMS. The causative lesion was most frequently located in the pons. We found that psRLS was more often bilateral, and usually detected later in time. Lesions in both the pontine base and tegmentum (together) were associated with unilateral psPLMS, whereas lesions in the corona radiata and adjacent basal ganglia were associated with bilateral RLS. Lesions confined to the corona radiata resulted in either unilateral or bilateral RLS., Conclusions: The observed differences in the clinical and radiological features of psRLS and psPLMS suggest that the pathophysiologies of the two conditions are distinct. Further research is needed to understand the pathophysiologies of primary RLS and PLMS., (© 2016 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Excessive fragmentary myoclonus (EFM) is a polysomnographic finding registered by the surface electromyography (EMG) and characterized as a result of the muscle activity consisting of sudden, isolated, arrhythmic, asynchronous and asymmetric brief twitches. The EMG potentials are defined by the exact criteria in The International Classification of the Sleep Disorders, 3rd edition and they appear with high intensity in all sleep stages. Clinical significance of EFM is unclear. It was observed in combination with other diseases and features such as obstructive and central sleep apnea, narcolepsy, periodic limb movements, insomnia, neurodegenerative disorders and peripheral nerve dysfunction. Relation to such wide range of diseases supports the opinion that EFM is nor a specific sleep disorder nor a specific polysomnographic sign. The option that EFM is a normal variant has also not been ruled out so far.

Study Objectives: Periodic leg movements during sleep (PLMS) occur within a subject as a series with a remarkably stable period defined by the intermovement interval (IMI). Sometimes a non-PLMS movement occurs intervening between two PLMS. PLMS scoring rules totally ignore these intervening leg movements (iLM). This implicitly assumes an iLM results from a process sufficiently independent from the periodic process producing PLMS that it does not affect the periodicity of the surrounding PLMS. This study for the first time tests this basic assumption and explores characteristics of iLM as a potentially significant class of leg movements during sleep., Methods: Leg movements were analyzed from two nights of polysomnography recordings from 27 RLS patients and 22 controls using the validated MATPLM1.1 program. All periods (IMI) between PLMS containing an iLM were compared to the local PLMS period defined as the immediately preceding PLMS IMI using pairwise two-sided Wilcoxon sign-rank tests. Similarly, iLM were tested to see if they started a new PLMS series by having the same period as the subsequent PLMS., Results: The periods (IMIs) containing iLM were longer than the previous periods in RLS subjects, but not controls (p < .05). The periods beginning with the iLM were shorter than the subsequent periods in both RLS and controls (p < .05)., Conclusions: iLM as a separate type of LM distort PLMS periodicity and do not restart PLMS series. iLM end PLMS series., (© Sleep Research Society 2016. Published by Oxford University Press on behalf of the Sleep Research Society. All rights reserved. For permissions, please e-mail journals.permissions@oup.com.)

Introduction: Hypnic jerks (HJs) are sudden contractions of one or more body segments occurring mostly at sleep onset. They are highly sporadic and affect all ages and both sexes with prevalence between 60% and 70% in the general population., Study Objectives: This study describes the frequency and the neurophysiological characteristics of HJs in a population of patients with parkinsonism by means of nocturnal video-polysomnographic recordings., Methods: This is a prospective cohort study and is reported following the STROBE guidelines. We analyzed the clinical and video-polysomnographic data of the first 66 consecutive patients recruited in the ongoing prospective study "Bologna motor and non-motor Prospective study on Parkinsonism at onset" (BoProPark). Each patient underwent a full neurological workup including a whole-night video- polysomnography. Neurophysiological characteristics including the propagation patterns of the HJs were studied with an extended muscle montage polysomnography., Results: We recorded a total of 62 HJs in 16 patients out of 66 (24%). Sleep parameters were not statistically different between patients with and without HJs. All HJs were spontaneous and occurred randomly throughout the night. Electromyographic analysis showed that muscle activity arose from different muscles with no prevalence of one over the other and without any ordered propagation. No recurring motor pattern of the jerks was detected., Discussion and Conclusions: Our findings demonstrated that HJs are a frequent, underestimated, sleep-related motor phenomenon in patients with parkinsonism. As they may represent a further cause of sleep disruption and insomnia, HJs should be actively examined. Neurophysiological analysis suggests a subcortical origin of HJs as shown previously for a healthy subject., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Study Objectives: To examine the association between periodic limb movements in sleep (PLMS) and change in selected aspects of cognition in community-dwelling older men., Methods: We studied 2,636 older men without dementia who underwent in-home polysomnography with measurement of the periodic limb movement index (PLMI) and periodic limb movement arousal index (PLMAI) using piezoelectric sensors. Random-effects models and logistic regression were used to examine the association between PLMI, PLMAI, and 3- to 4-y change in cognition., Results: After multivariable adjustment, men with a high PLMI had greater decline on the Trail Making Test - Part B (P trend = 0.02); those with a PLMI ≥ 30 were 48% more likely (odds ratio = 1.48, 95% confidence interval = 1.05-2.07) to experience the development of significant cognitive impairment (≥ 1 SD above mean change). Further adjustment for sleep efficiency, nocturnal hypoxemia, or dopaminergic medication use and analysis among men without Parkinson disease (n = 2,607) showed similar findings. No significant association was found for PLMAI or for Modified Mini-Mental State Examination scores., Conclusions: Among older men without dementia, higher PLMS frequency was associated with greater decline in cognition, particularly in executive function., (© 2016 Associated Professional Sleep Societies, LLC.)

Objectives: This study aimed to examine relationship between periodic limb movements during sleep (PLMS) and incident atrial fibrillation/flutter (AF)., Methods: Prospective multicenter cohort (n = 2273: adjudicated AF group; n = 843: self-reported AF group) of community-dwelling men without prevalent AF were followed for an average of 8.3 years (adjudicated) and 6.5 years (self-reported). PLMS index (PLMI, <5 (ref), ≥5 to <30, ≥30) and PLM arousal index (PLMAI, <1 (ref), ≥1 to <5, ≥5) were measured by polysomnography. Incident adjudicated and self-reported AF were analyzed by Cox proportional hazards and logistic regression, respectively, and adjusted for age, clinic, race, body mass index (BMI), alcohol use, cholesterol level, cardiac medications, pacemaker, apnea-hypopnea index, renal function, and cardiac risk. The interaction of age and PLMS was examined., Results: In this primarily Caucasian (89.8%) cohort of older men (mean age 76.1 ± 5.5 years) with BMI of 27.2 ± 3.7, there were 261 cases (11.5%) of adjudicated and 85 cases (10.1%) of self-reported incident AF. In the overall cohort, PLMI and PLMAI were not associated with adjudicated or self-reported AF. There was some evidence of an interaction of age and PLMI (p = 0.08, adjudicated AF) and PLMAI (p ≤ 0.06, both outcomes). Among men aged ≥76 years, the highest PLMI tertile was at increased risk of adjudicated AF (≥30 vs. <5; hazard ratio (HR) = 1.63, 1.01-2.63) and the middle PLMAI tertile predicted increased risk of both outcomes (1 to <5 vs. <1; adjudicated, HR = 1.65, 1.05-2.58; self-reported HR = 5.76, 1.76-18.84). No such associations were found in men aged <76 years., Conclusions: Although PLMS do not predict AF incidence in the overall cohort, the findings suggest PLMS increases incident AF risk in the older subgroup., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Movement and behavior disorders developing during sleep or in the transitional state from sleep to wakefulness are mostly unfamiliar to Japanese neurologists, primarily because these disorders are objects of basic research. In addition, the patients with sleep related movement and behavior disorders (SRMBD) show no signs and symptoms in our consulting rooms, and cannot be identified without polysomnography (PSG) attended by trained sleep technologists. Although attended PSG is not widely available in Japan, some kinds of SRMBD ought to be clinical targets for neurologists, and they are the scope of this review.