Unrefreshing naps and sleep architecture during the multiple sleep latency test in idiopathic hypersomnia.

Patients with idiopathic hypersomnia frequently report having unrefreshing naps. However, whether they have abnormal sleep architecture during naps that may explain their unrefreshing aspect is unknown. We compared sleep architecture during short daytime naps in patients with idiopathic hypersomnia reporting unrefreshing and refreshing naps. One-hundred and thirty-four patients tested with one-night polysomnography, followed by an adapted version of the Multiple Sleep Latency Test with four naps, were included. They were asked about the refreshing aspect of their habitual naps during a clinical interview. They were classified as having objective (Multiple Sleep Latency Test ≤ 8 min) or subjective idiopathic hypersomnia (Multiple Sleep Latency Test > 8 min), and as presenting refreshing or unrefreshing naps. We tested Group differences (refreshing versus unrefreshing naps) on nap sleep architecture in the whole sample and for subjective and objective idiopathic hypersomnia subgroups separately using ANCOVAs. No Group effects were observed in the Multiple Sleep Latency Test architecture in the whole sample and in objective and subjective idiopathic hypersomnia subgroups. This study provides preliminary evidence that reporting unrefreshing naps is not associated with clinically significant findings in Multiple Sleep Latency Test sleep architecture in patients with idiopathic hypersomnia. Given that naps taken by patients with idiopathic hypersomnia are typically long, future studies should investigate longer daytime sleep episodes.

Sweet or Bland Dreams? Taste Loss in Isolated REM-Sleep Behavior Disorder and Parkinson's Disease.

BACKGROUND: Hyposmia and isolated REM sleep behavior disorder are well-established features of prodromal Parkinson's disease (PD). OBJECTIVES: The objective of the present study was to evaluate whether taste loss (reported in PD and possibly suggesting brain stem involvement) is present at the isolated REM sleep behavior disorder stage. METHODS: We assessed taste function using the Taste Strip Test (evaluating 4 concentrations of bitter, sweet, sour, and salty) in 44 participants with isolated REM sleep behavior disorder, 19 with PD, and 29 controls. All participants underwent video-polysomnography, standardized questionnaires, and clinical examination, including olfactory assessment. RESULTS: Participants with isolated REM sleep behavior disorder and PD had lower taste scores than controls. There was no difference between isolated REM sleep behavior disorder and PD cohorts, nor was there any correlation between taste and olfaction, age, disease duration, cognition, or autonomic function. CONCLUSION: This study demonstrates for the first time the presence of taste impairment in isolated REM sleep behavior disorder that is independent of olfactory dysfunction and comparable to participants with PD. © 2021 International Parkinson and Movement Disorder Society.

Clinical Reasoning: A 3-Year-Old Boy With Abnormal Movements During Sleep.

We report a case of a 3-year-old boy who presented with abnormal movements that initially occurred only during sleep. Three years later, he went on to develop hyperkinetic movements during the daytime while awake. There was a strong family history of various paroxysmal neurologic disorders. In this report, we discuss the clinical approach, differential diagnosis, investigation, and treatment options for nocturnal hyperkinetic movements and paroxysmal movement disorders.

Are unrefreshing naps associated with nocturnal sleep architecture specificities in idiopathic hypersomnia?

STUDY OBJECTIVES: Unrefreshing naps are supportive clinical features of idiopathic hypersomnia (IH) and are reported by more than 50% of IH patients. They are, however, not mandatory for the diagnosis, and their pathophysiological nature is not understood. This study aimed at verifying whether IH patients with and without unrefreshing naps constitute two subtypes of IH based on their demographic/clinical characteristics, and sleep architecture. METHODS: One hundred twelve IH patients underwent a polysomnography (PSG) followed by a multiple sleep latency test (MSLT). They completed questionnaires on excessive daytime sleepiness, mood, and sleep quality. They were met by sleep medicine physicians who conducted a semi-structured clinical interview and questioned them on refreshing aspects of their naps. Patients who reported unrefreshing naps were compared to patients reporting refreshing naps on questionnaires, MSLT and PSG variables, with age as a covariable. As sensitivity analyses, we performed the same comparisons in participants presenting objective markers of IH and those diagnosed with IH based only on clinical judgment (subjective IH), separately. RESULTS: In the whole sample, 61% of patients reported unrefreshing naps. These participants had less awakenings, a lower percentage of N1 sleep, less sleep stage transitions, and a higher percentage of REM sleep on the nighttime PSG compared to the refreshing naps subgroup. When subjective and objective IH patients were tested separately, more group differences were observed on PSG for subjective IH patients. CONCLUSIONS: Patients with unrefreshing naps have less fragmented sleep compared to those with refreshing naps. Future studies should investigate whether this group difference indicates a weaker arousal drive.

The dynamics of emotional behaviors in rapid eye movement sleep.

Dream's emotions could exert a major role in desensitizing negative emotions. Studying emotional dynamics (how emotions fluctuate across time) during rapid eye movement (REM) sleep could provide some insight into this function. However, studies so far have been limited to dream reports. To bypass this limit, REM sleep behavior disorder (RBD), in which participants enact their dreams, enables direct access to overt emotional dream behaviors (such as facial expressions and speeches). In total, 17 participants with RBD, and 39.7 h of REM sleep video were analyzed. The frequency of emotional behaviors did not differ between REM sleep episodes of early and late night. Within individual REM sleep episodes, emotional behaviors exhibited a biphasic temporal course, including an increased frequency for the first 10 min, followed by a progressive decrease. The negative emotional behaviors occurred earlier (mean time: 11.3 ± 10 min) than positive (14.4 ± 10.7 min) and neutral behaviors (16.4 ± 11.8 min). Emotional behaviors of opposing (negative and positive) valences were observed in 31% (N = 14) of episodes containing at least one emotional behavior, and were separated by a median time of 4.2 [1.1-10.9] min. The biphasic temporal course of behaviors in REM sleep could include the generation reactivation of emotional content during the ascending phase, followed by processing and extinction during the descending phase. The earlier occurrence time of negative emotional behavior suggests that negative emotions may need to be processed first. The rapid succession of emotions of opposite valence could prevent prolonged periods of negative emotions and eventually nightmares.

Hypersomnia and narcolepsy in 42 adult patients with craniopharyngioma.

STUDY OBJECTIVES: To evaluate sleep, sleepiness, and excessive need for sleep in patients with craniopharyngioma (a suprasellar tumor which can affect sleep-wake systems). METHODS: A retrospective study of all adult patients living with craniopharyngioma referred to the sleep clinic, who received a sleep interview, nocturnal polysomnography, multiple sleep latency tests (MSLT), and 18-h bed rest polysomnography. Their sleep measurements were compared with those of age- and sex-matched healthy controls. RESULTS: Of 54 patients screened with craniopharyngioma, 42 were analyzed, 80% of whom complained of excessive daytime sleepiness. Sleep testing revealed that 6 (14.3%) of them had secondary narcolepsy (including one with cataplexy), and 11 (26.2%) had central hypersomnia associated with a medical disorder. Compared with controls, patients were more frequently obese, had a shorter mean sleep latency on MSLT, and slept longer on the first night. There was a nonsignificant trend for patients with (vs. without) narcolepsy and hypersomnia to be younger, to have a higher body mass index, to be more likely to have received radiation therapy, and to have more severe damage to the hypothalamus after surgery. Treatment with stimulants (modafinil, pitolisant, and methylphenidate) was beneficial in 9/10 patients. CONCLUSIONS: Nearly half of the patients with craniopharyngioma and sleep disorders have a central disorder of hypersomnolence (narcolepsy and hypersomnia), which should be investigated and lead to considerations beyond sleep apnea syndrome in these obese patients.

Eye movement patterns correlate with overt emotional behaviours in rapid eye movement sleep.

Growing evidence suggests that sleep plays a key role in regulating emotions. Rapid eye movements (REMs) in REM sleep could be associated with dreams emotions, but supporting evidence is indirect. To highlight this association, we studied the REM sleep during video-polysomnography of 20 subjects with REM sleep behaviour disorder (RBD), a model of enacted dreams offering direct access to the emotional content of the sleeper (face expression, speeches, behaviour). Video and the electro-oculography recordings were divided into 3 s time intervals and classified as non-behavioural, or behavioural (neutral, positive or negative emotions), and as containing no eye movements (EMs), slow eye movements (SEMs) or REMs (isolated or bursts). Compared to the absence of EMs, neutral behaviours successively increased in the presence of SEMs (odd ratio, OR = 1.4), then isolated REMs (OR = 2.8) and then REM bursts (OR = 4.6). Positive behaviours increased with SEMs (OR = 2.8) but did not increase further with isolated REMs (OR = 2.8) and REM bursts (OR = 3). Negative behaviours were absent with SEMs, increased with isolated REMs (OR = 2.6) and further with REM bursts (OR = 10.1). These results support an association between REMs and SEMs, and dream emotions.

Sleeping through a pandemic: impact of COVID-19-related restrictions on narcolepsy and idiopathic hypersomnia.

STUDY OBJECTIVES: To assess the impact of coronavirus disease 2019 (COVID-19)-related restrictions on narcolepsy type 1 (NT2), narcolepsy type 2 (NT2), and idiopathic hypersomnia (IH). METHODS: Participants with NT1, NT2, and IH followed in a university hospital completed an online 78-question survey assessing demographic, clinical, and occupational features of the population during the first COVID-19-related lockdown. RESULTS: A total of 219 of 851 (25.7%) respondents of the survey reported a mean increase of 1.2 ± 1.9 hours (P < .001) in night sleep time and a mean decrease of 1.0 ± 3.4 points (P < .001) on the Epworth Sleepiness Scale during lockdown. Bedtime was delayed by 46.1% of participants and wakeup time was delayed by 59.6%, driven primarily by participants with IH. Teleworkers (but not in-person workers) reported a mean increase of 0.9 ± 1.2 hours in night sleep (P < .001) and a mean decrease in sleepiness score of 1.6 ± 3.1 (P < .001). Cataplexy improved in 54.1% of participants with NT1. Sleepiness correlated with psychological wellness (r = .3, P < .001). As many as 42.5% enjoyed the lockdown, thanks to reallocation of time usually spent commuting toward longer sleep time, hobbies, and family time, and appreciated a freer napping schedule. Conversely, 13.2% disliked the lockdown, feeling isolation and psychological distress. CONCLUSIONS: Extended sleep time, circadian delay (in patients with IH), and teleworking resulted in decreased symptoms of central hypersomnias. These findings suggest that people with IH, NT1, and NT2 may benefit from a decrease in social and professional constraints on sleep-wake habits, and support advocacy efforts aimed at facilitating workplace and schedule accommodations for this population. CITATION: Nigam M, Hippolyte A, Dodet P, et al. Sleeping through a pandemic: impact of COVID-19-related restrictions on narcolepsy and idiopathic hypersomnia. J Clin Sleep Med. 2022;18(1):255-263.

Successful treatment of drug-resistant cataplexy with the anticholinergic drug tropatepine.

NONE: Narcolepsy type 1 is characterized by excessive daytime sleepiness and cataplexy, as well as hypocretin deficiency. Cataplexy (the loss of voluntary postural muscle tone, often in response to emotional stimuli) is one of the most disabling features and is associated with significant social impairment and risk of injury. Cataplexy is usually alleviated by antidepressants sodium oxybate and pitolisant. In this case report, we describe three patients with severe, drug-resistant cataplexy who experienced a dramatic improvement when treated with tropatepine, an anticholinergic muscarinic antagonist (commonly used to prevent neuroleptic-induced parkinsonism) after the usual treatments had failed. The single side effect was mild mouth dryness. In addition to providing a new therapeutic option for resistant cataplexy, this benefit supports a role of cholinergic muscarinic transmission in rapid eye movement sleep atonia.

Circadian Motor Improvement in MS and Core Body Temperature Dipping.

This case report describes a man with multiple sclerosis and insomnia who noticed improved motor function during nocturnal awakenings compared with daytime.

Successful Treatment of Somnambulism With OROS-Methylphenidate.

None: Somnambulism is a non-rapid eye movement sleep parasomnia with potential for significant injury as well as functional nighttime and daytime impairment. Clonazepam is frequently used as first line pharmacotherapy. However, the optimal treatment of somnambulism has not been established. In this article, we present the cases of two patients with severe somnambulism who showed a significant therapeutic response to osmotic release oral system methylphenidate (OROS-MPH). In addition to its practical therapeutic implications, this first report of the successful treatment of somnambulism with OROS-MPH may provide additional insight into the neurobiological underpinnings of this medical condition.