Search

To investigate clinical and video-polysomnography (VPSG) findings of hallucinatory experiences in patients suffering from disorders of arousal (DOA) in the absence of other pathologies.The authors retrospectively reviewed the records of 370 adults with DOA. Thirty (8.1%) patients concomitantly reported complex nocturnal visual hallucinations. VPSG recordings were scrutinized, and motor behavioral and electroencephalogram (EEG) patterns were classified according to previous descriptions of DOA.Thirty DOA patients reported seeing images of objects, people, and animals; either distorted, static, or mobile. The images disappeared with increased illumination in 80% of patients, and 23.3% reported preceding dream imagery. In addition to the classical DOA patterns on VPSG, a distinct pattern of behavioral and EEG manifestation associated with complex hallucinatory episodes was identified in 16 (53.3%) DOA patients. This consisted of low-voltage mixed-frequency EEG activity before eye opening that persisted while patients were observed staring or visually tracking before the onset of motor behavior.A novel, distinct behavioral and EEG pattern in patients with DOA and history of reported complex nocturnal visual hallucinations was identified. This may represent a unique phenotype of dissociation between sleep states that merits further investigation.

Background: Obstructive sleep apnea (OSA) is a chronic, highly prevalent, multi-system and sleep disorder, which may contribute to cognitive impairment and a variety of structural and neurophysiologic changes. The focus on OSA is warranted given its recognized links with major psychiatric and neurologic disorders, including Alzheimer's disease. Some preliminary studies suggest a dual effect of the inflammatory response in OSA. Neuroinflammation may present with initial, potentially adaptive and homeostatic, and later, a more distinctly maladaptive, precipitating and perpetuating role. ------ Objective: We here propose and argue in favour of the inflammatory process in the brain as a likely binding mechanism behind at least some effects that OSA may have on the brain and its function. Several OSA-triggered molecular and cellular events, that could lead to a neurodegenerative cascade, are similarly discussed. ----- Methods: This perspective reviews the body of literature that investigates potential links between the inflammatory processes in the brain and the OSA. A special emphasis is placed on a potential role for neuroplastin, a novel transmembrane synaptic protein involved in the neuroplasticity and known to be differentially regulated in the OSA. ----- Conclusions: The intricate interplay between neuroinflammation and other mechanistic correlates of OSA add to the evidence that neuroinflammation may be a key target for future therapeutic strategies in a number of comorbid disorders. The future studies will need to answer whether it is sleep fragmentation (SF) or intermittent hypoxia (IH) which may drive any such neuroinflammation.

IntroductionObstructive sleep apnoea (OSA) is a multisystem, debilitating, chronic disorder of breathing during sleep, resulting in a relatively consistent pattern of cognitive deficits. More recently, it has been argued that those cognitive deficits, especially in middle-aged patients, may be driven by cardiovascular and metabolic comorbidities, rather than by distinct OSA-processes, such as are for example ensuing nocturnal intermittent hypoxaemia, oxidative stress, neuroinflammation, and sleep fragmentation.MethodsThus, we undertook to define cognitive performance in a group of 27 middle-aged male patients with untreated OSA, who had no concomitant comorbidities, compared with seven matched controls (AHI mean ± S.D.: 1.9 ± 1.4 events/h; mean age 34.0 ± 9.3 years; mean BMI 23.8 ± 2.3 kg/m2). Of the 27 patients, 16 had mild OSA (AHI mean ± S.D.:11.7 ± 4.0 events/h; mean age 42.6 ± 8.2 years; mean BMI 26.7 ± 4.1 kg/m2), and 11 severe OSA (AHI 41.8 ± 20.7 events/h; age: 46.9 ± 10.9 years, BMI: 28.0 ± 3.2 kg/m2).ResultsIn our patient cohort, we demonstrate poorer executive-functioning, visuospatial memory, and deficits in vigilance sustained attention, psychomotor and impulse control. Remarkably, we also report, for the first time, effects on social cognition in this group of male, middle-aged OSA patients.ConclusionOur findings suggest that distinct, OSA-driven processes may be sufficient for cognitive changes to occur as early as in middle age, in otherwise healthy individuals.

Rapid eye movement (REM) sleep behaviour disorder (RBD) is a parasomnia characterised by abnormal behaviours arising during REM sleep. Described for the first time in humans just over 30 years ago, RBD is a condition of increasing clinical and research interest. Two forms of RBD have been identified: idiopathic (primary) and symptomatic (secondary). Idiopathic RBD may be the initial form of presentation of a neurodegenerative disorder. The secondary form of RBD is associated with an established neurological disorders, such as Parkinson’s disease (PD), dementia with Lewy bodies (DLB), multisystem atrophy (MSA) or narcolepsy. The diagnosis of RBD can often be straightforward, but does require confirmation by polysomnography to avoid misdiagnosis. The diagnosis is important, due to potential risks to the patient and their bedpartner, but also because of prognostic implications.

Background: The COVID-19 pandemic has led to significant changes in daily routines and lifestyle worldwide and mental health issues have emerged as a consequence. We aimed to assess the presence of sleep disturbances during the lockdown in the general population. Methods: Cross-sectional, online survey-based study on adults living through the COVID-19 pandemic. The questionnaire included demographics and specific questions assessing the impact of the pandemic/ lockdown on sleep, daytime functioning and mental health in the general population. Identification of sleep pattern changes and specific sleep-related symptoms was the primary outcome, and secondary outcomes involved identifying sleep disturbances for predefined cohorts (participants reporting impact on mental health, self-isolation, keyworker status, suspected COVID-19 or ongoing COVID-19 symptoms). Results: In total, 843 participants were included in the analysis. The majority were female (67.4%), middle aged [52 years (40–63 years)], white (92.2%) and overweight to obese [BMI 29.4 kg/m2 (24.1–35.5 kg/m2)]; 69.4% reported a change in their sleep pattern, less than half (44.7%) had refreshing sleep, and 45.6% were sleepier than before the lockdown; 33.9% had to self-isolate, 65.2% reported an impact on their mental health and 25.9% were drinking more alcohol during the lockdown. More frequently reported observations specific to sleep were ‘disrupted sleep’ (42.3%), ‘falling asleep unintentionally’ (35.2%), ‘difficulties falling’/‘staying asleep’ (30.9% and 30.8%, respectively) and ‘later bedtimes’ (30.0%). Respondents with suspected COVID-19 had more nightmares and abnormal sleep rhythms. An impact on mental health was strongly associated with sleep-related alterations. Conclusions: Sleep disturbances have affected a substantial proportion of the general population during the COVID-19 pandemic lockdown. These are significantly associated with a self-assessed impact on mental health, but may also be related to suspected COVID-19 status, changes in habits and self-isolation.

Idiopathic intracranial hypertension (IIH) is a condition of raised intracranial pressure (ICP). Obstructive sleep apnoea (OSA) has been shown to cause episodic rises in ICP and is frequently reported in patients with IIH. The aim of this study is to identify the prevalence of OSA in a cohort of IIH patients.We conducted a retrospective case notes review as part of a service evaluation of newly diagnosed IIH patients who were all referred for OSA screening with overnight pulse oximetry. The 3% oxygen desaturation index (3% ODI) was used to evaluate the presence and severity of OSA. The clinical outcomes of patients who received continuous positive airway pressure (CPAP) therapy as treatment for OSA were reviewed.In our cohort of newly diagnosed IIH patients, the yield of overnight pulse oximetry as a screening tool was 48.6% for OSA and 15.3% for moderate to severe OSA. We found that age (p = 0.0008), BMI (p 0.0001), vitamin BWe report the largest series of consecutive IIH patients screened for OSA using overnight pulse oximetry. The high rate of OSA highlights a potential role for the recognition and management of OSA in the IIH patient cohort. Further studies on the potential contribution of OSA as a cause of raised ICP in the IIH cohort is warranted.

Unlike sleep-walkers, patients with rapid-eye-movement-behaviour disorder (RBD) rarely leave the bed during the re-enactment of their dreams. RBD movements may be independent of spatial co-ordinates of the ‘outside-world’, and instead rely on (allocentric) brain-generated virtual space-maps, as evident by patients’ limited truncal/axial movements. To confirm this, a semiology analysis of video-polysomnography records of 38 RBD patients was undertaken and paradoxically restricted truncal/thoraco-lumbar movements during complex dream re-enactments demonstrated.

Obstructive sleep apnea is linked to cardiovascular disease, metabolic disorders and dementia. The precise nature of the association between respiratory events in obstructive sleep apnea, cortical or subcortical arousals, and cognitive, autonomic and oxidative stress consequences remains incompletely elucidated. Previous studies have aimed to understand the relationship between obstructive sleep apnea and arousal patterns, as defined by the cyclic alternating pattern, but results have been inconsistent, in part likely due to the presence of associated comorbidities. To better define this relationship, we analysed cyclic alternating patterns in patients with obstructive sleep apnea without any additional comorbidities. We identified 18 adult male, non-obese subjects with obstructive sleep apnea and no other comorbidities or medication history, who underwent whole-night electroencephalography and polysomnography. Cyclic alternating pattern analysis was performed and verified by certified somnologists. Pairwise linear regression analysis demonstrated an inverse relationship between obstructive sleep apnea severity and cyclic alternating pattern subtype A1, and a direct correlation with cyclic alternating pattern subtype A3. Cyclic alternating pattern subtypes A1 prevail in milder obstructive sleep apnea phenotype, whilst cyclic alternating pattern subtypes A2 and A3 overcome among moderate-to-severe obstructive sleep apnea patients. The milder obstructive sleep apnea group also presented higher sleep efficiency, and increased percentages of non-rapid eye movement stage 3 and rapid eye movement sleep, as well as longer cyclic alternating pattern sequences in N3, while severe obstructive sleep apnea patients spent more time in lighter sleep stages. These results imply/suggest a balance between cyclic alternating pattern's adaptive and maladaptive arousal processes in obstructive sleep apnea of differing severities. In milder obstructive sleep apnea (apnea-hypopnea index < 20), sleep continuity may be reinforced by cyclic alternating pattern subtype A1, whereas in more severe obstructive sleep apnea, decompensation of these sleep-stabilizing mechanisms may occur and more intrusive cyclic alternating pattern fluctuations disrupt sleep circuitry.

Summary Early studies posited a relationship between sleep and the basal ganglia, but this relationship has received little attention recently. It is timely to revisit this relationship, given new insights into the functional anatomy of the basal ganglia and the physiology of sleep, which has been made possible by modern techniques such as chemogenetic and optogenetic mapping of neural circuits in rodents and intracranial recording, functional imaging, and a better understanding of human sleep disorders. We discuss the functional anatomy of the basal ganglia, and review evidence implicating their role in sleep. Whilst these studies are in their infancy, we suggest that the basal ganglia may play an integral role in the sleep-wake cycle, specifically by contributing to a thalamo-cortical-basal ganglia oscillatory network in slow-wave sleep which facilitates neural plasticity, and an active state during REM sleep which enables the enactment of cognitive and emotional networks. A better understanding of sleep mechanisms may pave the way for more effective neuromodulation strategies for sleep and basal ganglia disorders.

Unpleasant dreamlike mentation can occur during non-rapid eye movement parasomnias, leading to associated panic attacks. The mentations are rarely remembered and are likely underreported. However, they may lead to significant personal distress and, if not addressed, may contribute to poorer clinical outcomes. Cotard le délire de negation are very rare nihilistic delusions, historically described with psychotic disorders. Their association with a variety of neurologic disorders, including migraine and cluster-headache, has also been reported. Here we present three cases of Cotard parasomnia during which distinct states of consciousness defined by nihilistic ideation occurred. Patients described believing they are dead or dying, while unable to perceive or experience their bodies in whole, or in part, as their own. A source analysis of the electroencephalographic fingerprint of these mentations suggests right-hemispheric hypoactivity subsequent to confusional arousals. Mechanistically, an aberrant activation of two major intrinsic brain networks of wakefulness, the salience network and the default mode network, is argued. CITATION: Gnoni V, Higgins S, Nesbitt AD, et al. Cotard parasomnia: le délire de negation that occur during the sleep-wake dissociation? J Clin Sleep Med. 2020;16(6):971–976.

Introduction: Microgravity is known to alter various physiological variables in humans. However, changes in neuroanatomy, cognition, and sleep have not been significantly studied in the literature. The aim of the present study was to determine the effect of seven-days of supine unloading on a supersaturated saline-filled water bed (hyper-buoyancy floatation, HBF), a novel Earth-based microgravity analogue, on cognition and sleep. Materials and methods: Twelve healthy male subjects were subject to seven continuous days of HBF, during which subjects' (floatonauts') major cognitive domains (psychomotor speed, attention, memory, executive function, and social cognition) and sleep were monitored by CANTAB testing batteries and 64-lead EEG respectively. Ten control subjects underwent identical cognitive testing regime protocol for comparison. Results: The unloading period of seven days resulted in a significant (p< 0.05) impairment in psychomotor speed and spatial working memory. However, a significant improvement was recorded in several domains of verbal memory. Various physiological changes, including the altered sleep architecture were recorded in floatonauts. Seven days of exposure lead to overall decreased sleep time, decreased non-rapid eye movement 2 sleep stage (N2) duration and decrease in overall NREM duration. Conversely, an increase in mean REM duration, sleep latency, along with increases in height and spine length were demonstrated. Postural changes appeared negatively related to changes in NREM duration, and verbal memory performance. Conclusions: Overall, the results suggest several cognitive and physiological changes induced by an intervention period of only seven days of HBF. These are hypothesized to occur due to the changes in neuroanatomical homeostasis, cephalic body fluid shift, physical activity, and sleep architecture. Further studies are required to investigate potential mechanisms underlying the observed multiple domain cognitive changes and their possible reversal.

Limited treatment options are available for patients with central hypersomnia, and many patients continue to have significant symptoms despite use of established therapies. Pitolisant is a novel wakefulness-promoting agent which has been demonstrated to reduce sleepiness in treatment naive patients with narcolepsy. Little is known about the utility of Pitolisant therapy in patients whose symptoms are refractory to other available medications. We assessed outcomes in hypersomnia patients who were resistant to or intolerant of conventional stimulant therapy. Patients with objectively confirmed central hypersomnia, who had persistent EDS despite use of ≥2 wakefulness promoting agents, were commenced on Pitolisant via a pharmacist-led medication management clinic. We prospectively recorded the Epworth Sleepiness Scale (ESS), Pittsburgh Sleep Quality Index (PSQI), and quality of life scores (EQ-5D visual analogue scale) at initiation and after 12 weeks of Pitolisant therapy. For the purposes of this analysis we defined responders as those with a reduction in ESS of ≥2. A total of 38 patients were given Pitolisant (55% female; 66% NT1; age 41.2±13.5 years). 6 stopped treatment due to adverse effects (50% insomnia; 33% apparent allergic reaction). Among those who completed 12 weeks of therapy, ESS reduced from 18.4±4.1 to 15.5±5.1 (p=0.001), PSQI reduced from 10.5±3.4 to 8.4±4.5 (p=0.006), and EQ-5D improved from 52±22 to 63±19 (p=0.004). Of the cohort, 17 (45%) were defined as responders – no significant differences in demographic factors, underlying diagnosis, or baseline symptom burden were observed between responders and non-responders. In a cohort of patients with difficult to treat central hypersomnia, Pitolisant therapy led to a reduction in daytime sleepiness and improved quality of life scores in a significant proportion of patients. Pitolisant may be a useful adjuvant therapy in some patients with treatment refractory hypersomnia.

Introduction Excessive daytime sleepiness (EDS) is disabling and it’s control is usually contingent on ongoing pharmacological therapy including: modafinil, methylphenidate, dexamfetamine, pitolisant, and sodium oxybate. Irrespective of the medication used, all pharmacological options must be taken to have an effect and while assessment of treatment adherence is standard clinical practice in many chronic conditions, in sleep medicine, evidence regarding adherence to prescribed medications is strikingly limited. The aim of this study was to assess degree and predictors of adherence to prescribed treatment in patients with narcolepsy attending a tertiary Sleep Disorders Centre. Methods We examined adherence to treatment in consecutive adult patients with a final diagnosis of narcolepsy by comparing prescription collection rates with prescribed therapy over a one-year period. Three levels of adherence were defined depending on the medication supplied in the last year in proportion to the total prescribed: poor (≤50%), intermediate (51–79%), and good (≥80%) adherence. Patients with adherence Results Demographic and clinical characteristics 162 patients were identified, from which 123 subjects with accurate information regarding current treatment regimen and adherence were included (see table 1). Good adherence was seen in 52.8% of patients, whilst 12.2% were intermediately and 35% poorly adherent. No difference was seen in proportion of good adherence between patients with refractory vs non-refractory symptoms (41.5% vs 58.5%; p=0.68). The proportion of refractory patients with suboptimal adherence was 44.9%. Patients with NT1 were less likely than those with NT2 to have suboptimal adherence (40.4% vs 75%; p=0.002). Discussion Our findings suggest that poor adherence is observed in a high proportion of patients with narcolepsy, and that a diagnosis of NT2 seems to be associated with non-adherence to prescribed treatment. Adherence to treatment should be routinely assessed in narcolepsy, particularly prior to initiating any step-up in therapy.

Although video polysomnography (vPSG) is not routinely recommended for the evaluation of typical cases of non-rapid eye movement (NREM) parasomnias, it can aid diagnosis of unusual cases, other sleep disorders and complicated cases with REM behaviour disorder (RBD), and in differentiating parasomnias from epilepsy. In this study, we aimed to assess vPSG findings in consecutive patients with a clinical diagnosis of NREM-parasomnia covering the whole phenotypic spectrum. Five hundred and twelve patients with a final diagnosis of NREM parasomnia who had undergone vPSG were retrospectively identified. vPSGs were analysed for features of NREM parasomnia and for the presence of other sleep disorders. Two hundred and six (40.0%) patients were clinically diagnosed with sleepwalking, 72 (14.1%) with sleep terrors, 39 (7.6%) with confusional arousals, 15 (2.9%) with sexsomnia, seven (1.4%) with sleep-related eating disorder, 122 (23.8%) with mixed phenotype, and 51 (10.0%) with parasomnia overlap disorder (POD). The vPSG supported the diagnosis of NREM parasomnia in 64.4% of the patients and of POD in 98%. In 28.9% of the patients, obstructive sleep apnea (OSA) or/and periodic limb movements during sleep (PLMS) were identified, most commonly in older, male, sleepy and obese patients. vPSG has a high diagnostic yield in patients with NREM parasomnia and should be routinely performed when there is diagnostic doubt, or in patients where there is a suspicion of OSA and PLMS.

The aim of the present study was to explore the modifications of EEG power spectra and EEG connectivity of resting state (RS) condition in patients with post-traumatic stress disorder (PTSD). Seventeen patients and seventeen healthy subjects matched for age and gender were enrolled. EEG was recorded during 5 min of RS. EEG analysis was conducted by means of the standardized Low Resolution Electric Tomography software (sLORETA). In power spectra analysis PTSD patients showed a widespread increase of theta activity (4.5–7.5 Hz) in parietal lobes (Brodmann Area, BA 7, 4, 5, 40) and in frontal lobes (BA 6). In the connectivity analysis PTSD patients also showed increase of alpha connectivity (8–12.5 Hz) between the cortical areas explored by Pz-P4 electrode. Our results could reflect the alteration of memory systems and emotional processing consistently altered in PTSD patients.

PURPOSE Hyperventilation (HV) is a commonly used electroencephalogram activation method. METHODS We analyzed EEG recordings in 22 normal subjects and 22 patients with focal epilepsy of unknown cause. We selected segments before (PRE), during (HYPER), and 5 minutes after (POST) HV. To analyze the neural generators of EEG signal, we used standard low-resolution electromagnetic tomography (sLORETA software). We then computed EEG lagged coherence, an index of functional connectivity, between 19 regions of interest. A weighted graph was built for each band in every subject, and characteristic path length (L) and clustering coefficient (C) have been computed. Statistical comparisons were performed by means of analysis of variance (Group X Condition X Band) for mean lagged coherence, L and C. RESULTS Hyperventilation significantly increases EEG neural generators (P < 0.001); the effect is particularly evident in cingulate cortex. Functional connectivity was increased by HV in delta, theta, alpha, and beta bands in the Epileptic group (P < 0.01) and only in theta band in Control group. Intergroup analysis of mean lagged coherence, C and L, showed significant differences for Group (P < 0.001), Condition (P < 0.001), and Band (P < 0.001). Analysis of variance for L also showed significant interactions: Group X Condition (P = 0.003) and Group X Band (P < 0.001). CONCLUSIONS In our relatively small group of epileptic patients, HV is associated with activation of cingulate cortex; moreover, it modifies brain connectivity. The significant differences in mean lagged coherence, path length, and clustering coefficient permit to hypothesize that this activation method leads to different brain connectivity patterns in patients with epilepsy when compared with normal subjects. If confirmed by other studies involving larger populations, this analysis could become a diagnostic tool in epilepsy.

Fatigue and sleep disorders are frequently reported in patients affected by Multiple Sclerosis (MS) but the causes and the relationship are not yet fully understood. This study aimed at evaluating their prevalence, at determining the relationships between clinical findings of MS and the occurrence of sleep disorders and at investigating the relations between sleep disorders and fatigue.One hundred and two MS patients were enrolled in the study. They were analyzed on both their clinical features (type of MS, disease duration, clinical severity, type of treatment, presence of spinal demyelinating lesions) and specific scales scores (Expanded Disability Status Scale, Modified Fatigue Impact Scale - MFIS, Self-Administered Anxiety Scale - SAS, Beck's Depression Inventory - BDI, Pittsburgh Sleep Quality Index - PSQI, Epworth Sleepiness Scale - ESS, and the Berlin's questionnaire for Obstruction Sleep Apnea Syndrome - OSAS).Patients with poor sleep quality are more frequently fatigued (p=0.001), have higher MFIS global scores (p0.001), higher prevalence of RLS symptoms (p=0.049), and show higher scores at BDI (p=0.017) and SAS (p≤0.001). Conversely patients with fatigue show older age (p=0.005), higher prevalence of sleepiness (p=0.021), higher prevalence of RLS symptoms (p=0.030), higher prevalence of poor sleep quality (p0.001) with higher PSQI scores (p0.001), higher scores on the BDI (p0.001) and SAS (p≤0.001).This study shows that MS is associated with a high prevalence of sleep complaints, including subjectively poor sleep quality, excessive daytime sleepiness, RLS and symptoms of OSAS. Further, it demonstrated a strict relation between fatigue and sleep disorders. Finally, it underlines their relationship with anxiety and depression in MS patients.

Pycnodysostosis (OMIM:265800) is an autosomal recessive genetic disorder due to a mutation in the cathepsin K gene, which causes a decrease of the bone turnover and a deterioration of the bone structure. Our aim was to describe a 5 year-old patient affected by pycnodysostosis, associated with an extremely severe obstructive sleep apnea syndrome, who was treated effectively with a combination of upper airway surgery and positive pressure ventilation. A 5 year-old child affected by pycnodysostosis was referred to us for failure to thrive, facial dysmorphisms and respiratory disorders, and who developed an extremely severe sleep apnea syndrome. Polysomnography showed extremely severe OSAS (AHI = 81.6 events/hour). The child was treated successfully with a combination of adenotonsillectomy, uvulo-palato-pharingo plasty (UPPP), followed by positive pressure ventilation. Polysomnographic recordings confirmed the striking reduction of obstructive respiratory events during sleep (from 81.6 to 12.3 events/hour). Lateral skull Rx and cephalometric measures showed that the Posterior Airway Space (PAS) increased from 3 to 19 mm. The decision to perform UPPP in association with adeno-tonsillectomy was motivated by the presence of palatal obstruction, caused by hypertrophic and prolapsed soft tissue. Our observations suggest that a conservative surgical treatment, consisting of adenotonsillectomy plus UPPP, may increases the patency of the upper airway, both at palatal and pharyngeal level. The combination of adenotonsillectomy plus UPPP, followed by CPAP ventilation, may avoid tracheotomy in very severe OSAS patients.

We describe a case of Restless Legs Syndrome (RLS) associated to periodic limb movements (PLMs) in a woman with a cervical schwannoma. Neurological examination, laboratory tests, neurography and electromyography were unremarkable. Neuroimaging evidenced a schwannoma at C3 level that compressed the right ventral surface of the medulla. Somatosensory-evoked potentials showed absence of the N13 after right median nerve stimulation and reduction of amplitude of the same component after left nerve stimulation. A video-polysomnography documented PLMs with a marked prevalence of the right-sided movements. We believe that the cervical schwannoma played a role in the pathogenesis of RLS and of lateralized PLMs.

Study Objectives: To evaluate sleep modifi cations induced by chronic benzodiazepine (BDZ) abuse. Methods: Cohort study, comparison of sleep measures between BDZs abusers and controls. Drug Addiction Unit (Institute of Psychiatry) and Unit of Sleep Disorders (Institute of Neurology) of the Catholic University in Rome. Six outpatients affected by chronic BDZ abuse were enrolled, (4 men, 2 women, mean age 53.3 ± 14.8, range: 34-70 years); 55 healthy controls were also enrolled (23 men, 32 women, mean age 54.2 ± 13.0, range: 27-76 years). All patients underwent clinical evaluation, psychometric measures, ambulatory polysomnography, scoring of sleep macrostructure and microstructure (power spectral fastfrequency EEG arousal, cyclic alternating pattern [CAP]), and heart rate variability. Results: BDZ abusers had relevant modifi cation of sleep macrostructure and a marked reduction of fast-frequency EEG arousal in NREM (patients: 6.6 ± 3.7 events/h, controls 13.7 ± 4.9 events/h, U-test: 294, p = 0.002) and REM (patients: 8.4 ± 2.4 events/h, controls 13.3 ± 5.1 events/h, U-test: 264, p = 0.016), and of CAP rate (patients: 15.0 ± 8.6%, controls: 51.2% ± 12.1%, U-test: 325, p < 0.001). Discussion: BDZ abusers have reduction of arousals associated with increased number of nocturnal awakenings and severe impairment of sleep architecture. The effect of chronic BDZ abuse on sleep may be described as a severe impairment of arousal dynamics; the result is the inability to modulate levels of vigilance.

Pycnodysostosis is an autosomal recessive disorder due to a mutation in the cathepsin K gene, which causes a decrease of the bone turnover; a review of the literature suggests that pycnodysostosis is frequently associated with severe respiratory obstruction, which needs surgical treatment. The aim of this paper is to describe the surgical treatment of a 3½-year-old girl affected by Pycnodysostosis complicated by a severe sleep-related respiratory disorder. The surgical treatment, consisting of adenotonsillectomy and palatoplasty, resulted in a striking amelioration of respiratory parameters and increased posterior airway space, and allowed the patient to avoid tracheotomy while awaiting for maxillo-mandibular surgery.

In this study, we evaluated cortical connectivity modifications by electroencephalography (EEG) lagged coherence analysis, in subjects with dissociative disorders and in controls, after retrieval of attachment memories. We asked thirteen patients with dissociative disorders and thirteen age- and sex-matched healthy controls to retrieve personal attachment-related autobiographical memories through adult attachment interviews (AAI). EEG was recorded in the closed eyes resting state before and after the AAI. EEG lagged coherence before and after AAI was compared in all subjects. In the control group, memories of attachment promoted a widespread increase in EEG connectivity, in particular in the high-frequency EEG bands. Compared to controls, dissociative patients did not show an increase in EEG connectivity after the AAI. Conclusions: These results shed light on the neurophysiology of the disintegrative effect of retrieval of traumatic attachment memories in dissociative patients.

Chronic insomnia is highly prevalent in the general population, provoking personal distress and increased risk for psychiatric and medical disorders. Autonomic hyper-arousal could be a pathogenic mechanism of chronic primary insomnia. The aim of this study was to investigate autonomic activity in patients with chronic primary insomnia by means of heart rate variability (HRV) analysis. Eighty-five consecutive patients affected by chronic primary insomnia were enrolled (38 men and 47 women; mean age: 53.2 ± 13.6). Patients were compared with a control group composed of 55 healthy participants matched for age and gender (23 men and 32 women; mean age: 54.2 ± 13.9). Patients underwent an insomnia study protocol that included subjective sleep evaluation, psychometric measures, and home-based polysomnography with evaluation of HRV in wake before sleep, in all sleep stages, and in wake after final awakening. Patients showed modifications of heart rate and HRV parameters, consistent with increased sympathetic activity, while awake before sleep and during Stage-2 non-REM sleep. No significant differences between insomniacs and controls could be detected during slow-wave sleep, REM sleep, and post-sleep wake. These results are consistent with the hypothesis that autonomic hyper-arousal is a major pathogenic mechanism in primary insomnia, and confirm that this condition is associated with an increased cardiovascular risk.

Objectives: This is an observational study aimed to investigate the activity of autonomic nervous system during sleep in patients with sleep-related migraine. Methods: Eight consecutive migraineurs without aura were enrolled (6 women and 2 men), aged 30 to 62 years (mean 48.1 ± 9.3 years). Inclusion criteria were: high frequency of attacks (> 5 per month) and occurrence of more than 75% of the attacks during sleep causing an awakening. Patients were compared with a control group of 55 healthy subjects (23 men and 32 women, mean age 54.2 ± 13.0 years), and with a further control group of 8 age- and gender-matched healthy controls. Patient and controls underwent polysomnography and heart rate variability analysis. Results: A signifi cant reduction of the LF/HF ratio during N2 and N3 sleep stages was observed in migraineurs compared with controls. No differences in sleep macrostructure were observed; CAP time and CAP rate were lower in migraineurs than in controls. Conclusions: These fiindicate a peculiar modifi cation of the autonomic balance during sleep in sleep-related migraine. The reduction of LF/HF ratio in NREM sleep was observed in controls, but it was quantitatively much more evident in migraineurs. Changes in LF/HF could be consequent to an autonomic unbalance which could manifest selectively (or alternatively become more evident) during sleep. These fitogether with the reduction in CAP rate, could be an expression of reduced arousability during sleep in patients with sleep-related migraine. The simultaneous involvement of the autonomic, arousal, and pain systems might suggest involvement of the hypothalamic pathways.

The n-back task is widely used to investigate the neural basis of Working Memory (WM) processes. The principal aim of this study was to explore and compare the EEG power spectra during two n-back tests with different levels of difficulty (1-back vs. 3-back). Fourteen healthy subjects were enrolled (seven men and seven women, mean age 31.21 ± 7.05 years, range: 23–48). EEG was recorded while performing the N-back test, by means of 19 surface electrodes referred to joint mastoids. EEG analysis were conducted by means of the standardized Low Resolution brain Electric Tomography (sLORETA) software. The statistical comparison between EEG power spectra in the two conditions was performed using paired t-statistics on the coherence values after Fisher's z transformation available in the LORETA program package. The frequency bands considered were: delta (0.5–4 Hz); theta (4.5–7.5 Hz); alpha (8–12.5 Hz); beta (13–30 Hz); gamma (30.5–100 Hz). Significant changes occurred in the delta band: in the 3-back condition an increased delta power was localized in a brain region corresponding to the Brodmann Area (BA) 28 in the left posterior entorhinal cortex (T = 3.112; p < 0.05) and in the BA 35 in the left perirhinal cortex in the parahippocampal gyrus (T = 2.876; p < 0.05). No significant differences were observed in the right hemisphere and in the alpha, theta, beta, and gamma frequency bands. Our results indicate that the most prominent modification induced by the increased complexity of the task occur in the mesial left temporal lobe structures.

Objective To measure the presence of the alpha-sleep anomaly in facioscapulohumeral muscular dystrophy (FSHD) and to evaluate the association between the sleep electroencephalogram (EEG) pattern and the presence of musculoskeletal pain. Design Cross-sectional study. Setting Sleep laboratory. Subjects Fifty-five consecutive adult FSHD patients, 26 women and 29 men, age 49.6 ± 15.1 years (range 18–76). Interventions Questionnaires and polysomnography. Outcome Measures Patients were asked to indicate if in the 3 months before the sleep study they presented persisting or recurring musculoskeletal pain. Patients who reported pain were asked to fill in the Italian version of the Brief Pain Inventory and the McGill Pain questionnaire, and a 101-point visual analog scale (VAS) for pain intensity. Polysomnographic recordings were performed. EEG was analyzed by means of Fast Fourier Transform. Four power spectra bands (δ 0–4 Hz, θ 4–8 Hz, α 8–14 Hz, β 14–32 Hz) were computed. Sleep macrostructure parameters and alpha/delta EEG power ratio during non rapid eye movement (NREM) sleep were compared between patients with and without pain. Results Forty-two patients in our sample reported chronic pain. VAS mean score was 55.2 ± 23.8 (range 10–100), pain rating index score was 13.8 ± 10.2, and present pain intensity was 2.5 ± 0.8. The statistical analysis documented an increased occurrence of the alpha and beta rhythms during NREM sleep in FSHD patients with pain. Significant correlations were observed between the alpha/delta power ratio during NREM sleep and pain measures. Conclusions Chronic musculoskeletal pain is frequent in FSHD patients, and it represents a major mechanism of sleep disruption.

Objective The aim of the present study was to develop and validate a software tool for the detection of movements during sleep, based on the automated analysis of video recordings. This software is aimed to detect and quantify movements and to evaluate periods of sleep and wake. Methods We applied an open-source software, previously distributed on the web (Zoneminder, ZM), meant for video surveillance. A validation study was performed: computed movement analysis was compared with two standardised, ‘gold standard’ methods for the analysis of sleep–wake cycles: actigraphy and laboratory-based video-polysomnography. Results Sleep variables evaluated by ZM were not different from those measured by traditional sleep-scoring systems. Bland–Altman plots showed an overlap between the scores obtained with ZM, PSG and actigraphy, with a slight tendency of ZM to overestimate nocturnal awakenings. ZM showed a good degree of accuracy both with respect to PSG (79.9%) and actigraphy (83.1%); and had very high sensitivity (ZM vs. PSG: 90.4%; ZM vs. actigraphy: 89.5%) and relatively lower specificity (ZM vs. PSG: 42.3%; ZM vs. actigraphy: 65.4%). Conclusions The computer-assisted motion analysis is reliable and reproducible, and it can allow a reliable esteem of some sleep and wake parameters. The motion-based sleep analysis shows a trend to overestimate wakefulness. Significance The possibility to measure sleep from video recordings may be useful in those clinical and experimental conditions in which traditional PSG studies may not be performed.

Chiari malformation (CM) is the downward herniation of the caudal part of the cerebellum and/or medulla oblongata into the spinal canal. It can alter several neurological functions, including respiratory control and upper airway motility, and can be the cause of sleep-disordered breathing (SDB). The authors describe a 6-year-old boy affected by CM Type II associated with myelomeningocele who showed symptoms indicative of severe airway obstruction during sleep. Polysomnography revealed severe obstructive sleep apnea syndrome (OSAS). Magnetic resonance imaging demonstrated herniation of the cerebellar tonsils and diffuse ventricular dilation with a large pseudocystic formation in the third ventricle. Surgical marsupialization of the cystic wall was performed, associated with ventriculocystostomy and endoscopic replacement of the ventricular catheter. Polysomnography repeated 2 months after surgery revealed a striking improvement in the sleep-related respiratory pattern. The pathogenesis of OSAS was probably referable to a combination of CM and elevated intracranial pressure. However, the striking improvement of symptoms after ventriculoatrial shunt placement suggested that hydrocephalus plays a major role in this condition. Assessment and effective treatment of SDB is crucial in the care of patients with CM.

The purposes of the study are: (1) to establish if cephalometry and upper airway examination may provide tools for detecting facioscapulohumeral (FSHD) patients at risk for obstructive sleep apnea syndrome (OSAS); and (2) to correlate cephalometry and otorhinolaryngologic evaluation with clinical and polysomnographic features of FHSD patients with OSAS.Patients were 13 adults affected by genetically confirmed FSHD and OSAS, 11 men, with mean age 47.1 ± 12.8 years (range, 33-72 years). All underwent clinical evaluation, Manual Muscle Test, Clinical Severity Scale for FSHD, Epworth Sleepiness Scale, polysomnography, otorhinolaryngologic evaluation, and cephalometry.Cephalometric evidence of pharyngeal narrowing [posterior airways space (PAS) 10 mm] was present in only one patient. The mandibular planus and hyoid (MP-H) distance ranged from 6.5 to 33.1 mm (mean, 17.5 ± 7.8 mm). The mean length of soft palate (PNS-P) was 31.9 ± 4.8 mm (range, 22.2 to 39.7 mm). No patient presented an ANB angle 7°. There was no significant correlation between cephalometric measures, clinical scores, and PSG indexes. PAS and MP-H were not related to the severity of the disease.Upper airway morphological evaluation is of poor utility in the clinical assessment of FSHD patients and do not allow to predict the occurrence of sleep-related upper airway obstruction. This suggests that the pathogenesis of OSAS in FSHD is dependent on the muscular impairment, rather than to the anatomy of upper airways.