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Abstract: Restless genital syndrome refers to excessive and persistent sensations of genital and clitoral arousal with the absence of conscious feeling of sexual desire. These sensations have been linked to symptoms of restless legs syndrome (RLS) or an overactive bladder. Restless genital syndrome may be related to small fiber sensory neuropathy of the dorsal nerve of the clitoris, so it more frequently occurs in women. The association with RLS suggests a possible dopaminergic mechanism. We report a case of a 74-year-old woman who presented restless genital symptoms every night in association with RLS symptoms. During polysomnography she reported 3 awakenings from rapid eye movement sleep with associated genital symptoms followed by a period of insomnia with RLS symptoms and sexual activity. The frequency and the intensity of the symptoms were responsive to treatment by pramipexole after 1 week of treatment and the efficacy was maintained at follow-up., (© 2017 American Academy of Sleep Medicine)

Objectives: Autonomic nervous system dysfunction has been described in narcolepsy with cataplexy affecting sympathetic functions. In this study we analyzed whether altered diurnal and nocturnal cardiovascular control is present in idiopathic hypersomnia (IH)., Methods: Fourteen drug-free patients aged 26.2 ± 7 years and 14 age-matched controls were examined. Clinical data, 24-h polysomnography, heart rate (HR) variability, and the HR response to spontaneous arousal were available., Results: Sleep macrostructure was comparable between controls and patients, with the latter having significantly longer sleep time, a higher number of sleep cycles (p < 0.0001), and low sleep efficiency (p < 0.01). The HR variability indices did not differ between groups, except for the rise of high frequency (HF) and HFnu in patients (p < 0.05) associated with blunted sympathetic indices (p < 0.01). These parasympathetic alterations were present for light, slow wave, and rapid eye-movement sleep and persisted for all sleep cycles. Compared to controls, the HR arousal response was significantly higher (p < 0.01) in patients starting before the arousal onset and persisting into the post-arousal period., Conclusions: In IH patients a dysfunction of the parasympathetic activity during awake and sleep and an altered autonomic response to arousals are present. These findings suggest an impaired parasympathetic function that may explain some vegetative symptoms present in this type of central hypersomnia., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Objective: Several factors, such as homeostatic and circadian influences, may affect the density of cortical and subcortical arousals (AR). The purpose of this study was to examine the time-of-night and the first night effect on AR response., Methods: AR were classified into microarousals (MA), phases of transitory activation (PAT), delta (D-burst) and K-complex burst (K-burst). The AR density and duration was analyzed during two consecutive nights with the analysis of sleep stage and sleep cycle in thirty-six healthy subjects., Results: D- and K-burst showed a trend toward progressive decline across sleep cycles (p<0.0001). While MA rate was unaffected throughout sleep cycles, PAT index increased across the night (p=0.002). The density and duration of each group of AR exhibited reproducibility without significant differences between nights. An individual inter-night variability in AR density was found independently of night and sleep structure., Conclusions: While homeostatic and circadian influences affect nighttime subcortical and MA responses, a wakefulness drive modulates the occurrence of AR with movements. Although the pattern of AR responses was highly reliable from the first to second night, the substantial inter-individual variability suggests the existence of an individual susceptibility., Significance: The first night effect on arousal response is affected by individual susceptibility and circadian and homeostatic influences.

The maximal inspiratory effort recorded at the end of apnea has been considered as an index of arousal threshold in obstructive sleep apnea syndrome (OSAS). Previous investigations have shown that the arousal threshold is higher in patients with OSAS than in normal subjects. The aim of the present study was to investigate the effect of continuous positive airway pressure (CPAP) treatment on the inspiratory-effort-related arousal threshold in patients with OSAS. In ten male patients, 40 episodes of apnea during stage 2 non-REM (NREM) sleep were analyzed. Apnea duration (t), esophageal pressure (Pes) at the first occluded breath (Pes1), the minimum of the three initial Pes swings (Pes min), the maximum of the three final Pes swings (Pes Max), DeltaPes (Pes Max-Pes min), RPes (rate of increase of intrathoracic pressure, DeltaPes/t), n (number of occluded breaths during apnea), DeltaPes/n, n/t, and SaO(2) were determined before and after occlusion. These apneic episodes were compared to ten episodes of apnea provoked by a mask occlusion device after 1, 7, 30, and 90 days of CPAP treatment. The therapy resulted in a decrease in the inspiratory-effort-related arousal threshold, as measured by a reduction of Pes Max, without significant changes in apnea duration and apnea-related hypoxemia. Pes1 and DeltaPes/n, which are markers of respiratory drive, significantly decreased between observations. CPAP treatment decreases the inspiratory-effort-related arousal threshold and induces a decrease in ventilatory drive in response to upper airway occlusion.

Objective: Arousal (AR) from sleep is associated with an autonomic reflex activation raising blood pressure and heart rate (HR). Recent studies indicate that sleep deprivation may affect the autonomic system, contributing to high vascular risk. Since in sleep disorders a sleep fragmentation and a partial sleep deprivation occurs, it could be suggested that the cardiovascular effects observed at AR from sleep might be physiologically affected when associated with sleep deprivation. The aim of the study was to examine the effect of sleep deprivation on cardiac arousal response in healthy subjects., Methods: Seven healthy male subjects participated in a 64 h sleep deprivation protocol. Arousals were classified into four groups, i.e. >3<6 s, >6<10 s, >10<15 s and >15 s, according to their duration. Pre-AR HR values were measured during 10 beats preceding the AR onset, and the event-related HR fluctuations were calculated during the 20 beats following AR onset. As an index of cardiac activation, the ratio of highest HR in the post-AR period over the lowest recorded before AR (HR ratio) was calculated., Results: For AR lasting less than 10 s, the occurrence of AR induces typical HR oscillations in a bimodal pattern, tachycardia followed by bradycardia. For AR lasting more than 10 s, i.e. awakenings, the pattern was unimodal with a more marked and sustained HR rise. The HR response was consistently similar across nights, during NREM and REM sleep, without difference between conditions., Conclusions: Overall, total sleep deprivation appeared to have no substantial effect on cardiac response to spontaneous arousals and awakenings from sleep in healthy subjects. Further studies are needed to clarify the role of chronic sleep deprivation on cardiovascular risk in patients with sleep disorders., Significance: In healthy subjects acute prolonged sleep deprivation does not affect the cardiac response to arousal.

Study Objectives: The hierarchical definition of arousals from sleep includes a range of physiologic responses including microarousals, delta and K-complex bursts, and variations in autonomic system. Whether patterns in slow-wave electroencephalographic activity and autonomic activation are primary forms of arousal response can be addressed by studying effects of total sleep deprivation. We therefore examined changes in arousal density during recovery sleep in healthy subjects., Design: Participants spent 6 consecutive 24-hour periods in the laboratory. Nights 1 and 2 were baseline nights followed by 64-hour total sleep deprivation, then 2 consecutive recovery nights., Setting: Sleep-deprivation protocol was conducted under laboratory conditions with continuous behavioral and electrophysiologic monitoring., Participants: Twelve drug-free men aged 27.4 +/- 7.9 years were studied. None reported sleepiness or altered sleep-wake cycle, and none had neurologic, psychiatric or sleep disorders., Intervention: N/A., Measurements and Results: Arousals were classified into 4 levels: microarousals, phases of transitory activation, and delta and K-complex bursts. Sleep deprivation induced changes in the density of considered arousals except phases of transitory activation, with a distinct pattern among the different types. The greatest change was found for microarousals, which showed a significant decrease in the first recovery night (P = .01), with return to baseline thereafter. A fall in K-complex and delta-burst density was noted in the first recovery night, not, however, reaching statistical significance. The phases of transitory activation rate were virtually unaffected throughout the experimental nights., Conclusions: We conclude that homeostatic sleep processes exert an inhibitory effect on arousal response from sleep with a significant effect only on the microarousal density. Decreased delta and K-complex burst rates, though not significant, support the hypothesis that they may be activating processes, probably modulated by factors independent from those implicated in cortical arousal.

Objective: The temporal evolution of periodic leg movements (PLM) and the relationship of their arousing effect on sleep episode has not been extensively investigated. We studied the nocturnal evolution of PLM associated or not with microarousal (MA) and associated with slow wave activity (PLM with slow wave activity) in 23 patients with PLM and/or restless legs syndrome (RLS)., Methods: All subjects had PLM associated with MA or with slow wave activity as well as without MA and all slept for 4 sleep cycles. Spectral electroencephalogrpahic (EEG) analysis was done for the 4 sleep cycles to assess the nocturnal variation in slow wave activity (SWA)., Results: Sixty percent of PLM were associated with MA, 4% were associated with slow wave activity whereas 36% showed no EEG changes. There was a clear prevalence of PLM with MA in stages 1 and 2 while PLM without MA were prevalent in slow wave sleep. The night-time PLM index progressively declined from the first to the last sleep cycle (P<0.005), without differences between PLM types, or between PLM and RLS patients. The decline of PLM duplicated the temporal trend in SWA over consecutive sleep cycles., Conclusions: PLM showed a typical pattern of progressive decline throughout the night following the exponential decline in SWA. These over-time variations occurred independently of changes in the rate of PLM associated or not with MA or associated with slow wave activity, suggesting that variations in arousal threshold and sleep propensity did not affect the PLM arousing effect. The PLM-related arousal response might be affected by interaction of circadian and sleep stage influences with the addition of sleep oscillatory processes.

Objectives: A hierarchy in arousal response has been proposed for spontaneous arousal by analyzing the temporal changes in heart rate (HR) and electroencephalographic (EEG) activity. To address the question as to whether the same continuum may be proposed in sleep disorders, we performed temporal spectral EEG and HR analyses during periodic leg movements (PLM) associated or not with microarousal (MA)., Methods: Data were obtained in 12 patients with restless leg syndrome and/or PLM syndrome. PLMs were classified into 3 types including PLM associated with MA, PLM without MA, and PLM associated with delta or K-complex bursts. HR and EEG spectral analyses were done for 10s before and 10s after the PLM onset., Results: Each type of PLM was associated with a typical EEG and autonomic pattern consisting of an increase in HR and delta band activity before the PLM, regardless of the presence or absence of MA. Thereafter, a rise in delta, alpha and beta(2) activity was noted associated with tachycardia. This was greater when MA or bursts of slow wave activity were present. In the period following the PLM, HR, delta and alpha power showed a long-lasting decrease with values significantly below the baseline., Conclusions: From these data, we can conclude that: (1) cardiac and cerebral changes occur in association with PLM even when MA cannot be detected; (2) the combined increase in delta activity and HR before the onset of PLM suggests that these changes are part of the arousal response during PLM; (3) the graded arousal response during PLM confirms that the human arousal response involves a progression of central nervous system activation from brainstem to cortical level.

Objectives: One major subject of discussion in sleep studies is whether bursts of K-complexes (K-bursts) and delta waves (D-bursts), expressions of a subcortical arousal, truly reflect an arousal response during sleep. To address this question we studied the changes in heart rate (HR) during spontaneous arousals in healthy subjects., Methods: Twenty-seven healthy adults were examined. Arousals were graded in 4 levels, including the standard definition of a microarousal (MA), phases of transitory activation (PAT), D-bursts and K-bursts. HR was analyzed for 10 beats before and 20 beats during arousal. EEG spectral analysis was performed for all types of arousals, including in the analysis the 20 s period preceding the actual event., Results: Each type of arousal was associated with HR changes consisting of a tachycardia followed by a bradycardia. Changes were more pronounced during MA and PAT. Detailed analysis of the HR response showed that HR always increased before MA and PAT onset, associated with a rise in delta, theta and fast EEG activities, and suggesting a cerebral activation., Conclusions: Our data suggest that such subcortical arousals represent a real arousal response inducing cardiac activation similar to that found during MA and PAT. During MA and PAT, a rise in HR appears before the onset of the actual arousal associated with an increase in EEG slow and fast activity. The link between EEG and HR variation during MA and PAT and the fluctuations in HR during subcortical arousal suggest a continuous spectrum in the arousal mechanisms, starting at the brainstem level and progressing to cortical areas.

Objective: To investigate other physiologic changes that occur with periodic leg movements during sleep (PLMS) that might be considered to be more sensitive indices of sleep fragmentation., Background: Although PLMS are associated with recurrent microarousals (MA), the frequency of PLMS with MA does not correlate with objective daytime sleepiness. It is postulated that the lack of correlation results from the low sensitivity of the standard criteria used to score MA., Methods: Ten drug-free patients with a polygraphic and clinical diagnosis of restless legs syndrome (RLS) and PLMS were examined. The EEG correlates of PLMS were analyzed by visual scoring and spectral analysis during PLMS that ended in a visible microarousal (PLMS with MA) or not (PLMS without MA). The R-R interval in the EKG signal was also examined., Results: A total of 34% of PLMS were associated with MA lasting >3 seconds, and 3% of PLMS were associated with MA lasting <3 seconds. Although PLMS with MA were associated with an increase in alpha activity, for PLMS without MA a significant increase in delta and theta activity was present. Both types of PLMS induced a shortening of the R-R interval; this was particularly more marked for PLMS with MA., Conclusions: First, visual scoring of MA that include a duration of less than 3 seconds has little effect on the detection of PLMS with MA. Second, EEG activation and tachycardia are present during both types of PLMS. Third, a hierarchy in the arousal response is present-going from autonomic activation to bursts of delta activity to alpha activity to a full awakening.

It has recently been described that the maximal respiratory effort developed at the end of an apnea (Pesmax)--which is regarded as an index of arousal threshold in patients with obstructive sleep apnea syndrome (OSA)--increases progressively during the night, probably as a consequence of associated sleep fragmentation. In order to find out whether the nocturnal trend of Pesmax may be more influenced by a sleep-dependent circadian rhythm than by sleep fragmentation, we revised the polygraphic recordings of 37 patients in whom obstructive apneas were recorded for at least 7 hours. In 15 of these patients, analysis was made for eight hours of the night. During each hour we analyzed at least 7 obstructive apneas, in which we measured the minimal esophageal pressure at the start of the apnea, the maximum value recorded at the end of the apnea (Pesmax), the difference from the minimum to the maximum (delta Pes), and the rate of increase in esophageal pressure (RPes). As indices of sleep fragmentation, we defined the number of arousals, awakenings and sleep state transitions. In the group of patients as a whole, we found a trend toward a gradual increase for apnea duration (F = 98.8, p < 0.001) and Pesmax F = 31.6, p < 0.001) which was significant from the first to the last hour. The time-dependent evolution of apnea duration and Pesmax showed that the rise in these two variables peaked during the first 3 hours of sleep, followed by a plateau and a decrease in the last hour of the night. This temporal profile was more evident when the analysis was available for 8 hours. No significant changes across the night were found for nocturnal hypoxemia and number of arousals. Considering the slope of Pesmax changes across the night, we saw that neither the apnea+hypopnea index nor the indices of sleep fragmentation affected the nocturnal trend. The present data demonstrate the presence of a nocturnal trend in arousal threshold in OSA patients independent of sleep fragmentation. The biphasic evolution of the arousal threshold may be caused by factors that influence the circadian and homeostatic processes.

It has been recently described that the overnight increase in maximal end-apnoeic oesophageal pressure (P(oes,max)), considered as an index of the arousal threshold to occlusion, mostly contributes to apnoea lengthening during the night. However, the rise in apnoea duration could also be caused by changes in hypoxaemia, chemosensitivity and upper airway resistance. To better define the relative contributions of each of these factors, we examined the recordings of nine patients. Before apnoea, the mean pulmonary resistance at peak inspiratory flow (RPIF) was computed. During apnoea, all swings in oesophageal pressure (P(oes)) were measured to define the P(oes,max), the increase from the minimum to the maximum (deltaP(oes)), the rate of increase in P(oes) (RP(oes)) and the P(oes) at the first occluded breath (P(oes,1)). A gradual and significant increase in apnoea duration (p=0.02), P(oes,max) (p=0.02) and deltaP(oes) (p=0.006) was present across the night without any changes in oxygen saturation, RPIF, and P(oes,1). The slope of increase in P(oes,max), apnoea duration and deltaP(oes) was correlated with the apnoea/hypopnoea index. We conclude that in obstructive sleep apnoea, the nocturnal rise in apnoea duration is attributable more to an increase in the arousal threshold related to apnoea recurrence than to changes in chemosensitivity and upper airway resistance.

Six patients complained of distressing sudden awakenings with abnormal motor activity during sleep causing insomnia. Polysomnography showed paroxysmal short-lasting arousals during NREM, especially slow-wave sleep, associated with complex movements and autonomic activation. Ictal and interictal EEG never showed epileptic discharges except in 1 patient who also had a tonic-clonic seizure during sleep. Carbamazepine was the only effective medication in 2 patients. Paroxysmal arousals represent a sleep disturbance that may be related to deep epileptic foci.