Nocturnal agitation: From sleep state dissociation to sleep-related dissociative state.

Nocturnal agitation refers to a broad spectrum of symptoms from simple movements to aggressive behaviors with partial or complete loss of awareness. An accurate identification of its etiology is critical for appropriate therapeutic intervention. In children and young adults, distinguishing between non-rapid eye movement (NREM) sleep parasomnias and psychogenic non-parasomniac manifestations, a condition known as sleep-related dissociative disorder (SRDD), can be challenging. This review aims to summarize current clinical, neurophysiological, and epidemiological knowledge on NREM parasomnia and SRDD, and to present the pathophysiological hypotheses underlying these nocturnal manifestations. Sleepwalking, sleep terror and confusional arousals are the three main presentations of NREM parasomnias and share common clinical characteristics. Parasomniac episodes generally occur 30minutes to three hours after sleep-onset, they are usually short, lasting no more than few minutes and involve non-stereotyped, clumsy behaviors with frequent amnesia. The prevalence of NREM parasomnia decreases from 15-30% in children to 2-4% in adults. Parasomniac episodes are incomplete awakening from the deepest NREM sleep and are characterized by a dissociated brain activity, with a wake-like activation in motor and limbic structures and a preserved sleep in the fronto-parietal regions. SRDD is a less known condition characterized by dramatic, often very long episodes with frequent aggressive and potentially dangerous behaviors. SRDD episodes frequently occur in quiet wakefulness before falling asleep. These dissociative manifestations are frequently observed in the context of psychological trauma. The pathophysiology of SRDD is poorly understood but could involve transient changes in brain connectivity due to labile sleep-wake boundaries in predisposed individuals. We hypothesize that SRDD and NREM parasomnia are forms of sleep-related dissociative states favored by a sleep-wake state dissociation during sleep-onset and awakening process, respectively.

A challenging case of sexsomnia in an adolescent female presenting with depressive-like symptoms.

Sleep-related sexualized behaviors occur in the parasomnia known as sexsomnia, recognized as a variant of confusional arousals in the International Classification of Sleep Disorders, third edition. These instinctive behaviors of a sexual nature emerge from deep non-rapid eye movement sleep, and patients often present with distinguishing features within this sleep disorder category. There are often adverse psychosocial consequences and not uncommonly medicolegal implications. While associations to psychiatric consequences from the sexsomnia have been demonstrated and efforts to further typify this condition have been made, sexsomnia remains incompletely characterized in the more than 200 published cases to date, with male predominance. We now present the first reported case of an adolescent female with sexsomnia that was triggered by the onset of Crohn's disease and its treatment with azathioprine and with interpersonal consequences leading to an initial psychiatric consultation on account of depressive symptoms. These symptoms were deemed to be secondary to the sexsomnia. In addition to describing unusual and clinically relevant features in this case of sexsomnia, this original case provides insights into triggers, predisposing factors, perpetuating factors, and therapeutic considerations that are important for raising awareness in sleep clinicians, primary care providers, and mental health professionals. CITATION: Brás J, Schenck CH, Andrade R, et al. A challenging case of sexsomnia in an adolescent female presenting with depressive-like symptoms. J Clin Sleep Med. 2023;19(10):1845-1847.

Transient Central Sleep Apnea Runs Triggered by Disorder of Arousal in a Child.

ABSTRACT: We report the case of a 5-year-old girl with frequent nocturnal episodes of disorder of arousal (confusional arousals, sleep terrors, and sleep walking), occurring at the end of periods of slow wave sleep, followed by return to sleep accompanied by the occurrence of periodic breathing with a run of approximately 10 to 20 central events. The duration of the central events and oxyhemoglobin desaturation were both maximum at the beginning of each run and became progressively less prominent with the development of the sequences. Night episodes disappeared with bedtime clonazepam but behavioral problems occurred as a paradoxical response; thus, clonazepam was stopped. Sleep extension and melatonin were then started, which were followed by a reduction of night episode frequency and intensity. This observation appears to be the first report of central sleep apnea sequences triggered by parasomnia and, if confirmed by additional reports, it might be considered to be a possible new classification of "complex parasomnia."

An interprofessional approach to managing children with treatment-resistant enuresis: an educational review.

Enuresis (intermittent urinary incontinence during sleep in a child aged ≥ 5 years) is commonly seen in paediatric practice. Despite the availability of effective interventions, treatment resistance is encountered in up to 50% of children. In this educational review we attempt to provide insight into the causes of treatment resistance, and offer practical suggestions for addressing this condition using an interprofessional approach. We explore the pathophysiology of and standard treatments for enuresis and discuss why standard treatments may fail. An interprofessional approach to treatment resistance is proposed which utilises the expertise of professionals from different disciplines to address the problems and barriers to treatment. The two interprofessional approaches include a multidisciplinary approach that involves the patient being sent to experts in different disciplines at different times to address their treatment resistance utilising the skills of the respective experts, and an interdisciplinary approach that involves a patient being managed by members of interdisciplinary team who integrate their separate discipline perspectives into a single treatment plan. Although an interdisciplinary approach is ideal, interdisciplinary teams may not be available in all circumstances. Understanding the roles of other disciplines and engaging clinicians from other disciplines when appropriate can still be helpful when treatment resistance is encountered.

Why is a prone sleeping position dangerous for certain infants?

The prone (face down) sleeping position is known to be associated with a significantly increased risk of sudden and unexpected death in infancy (sudden infant death syndrome or SIDS), however, the reasons for this are unclear. Suggested mechanisms have involved suffocation from occlusion of the external airways by soft bedding/pillows or from flattening of the nose with backward displacement of the tongue, rebreathing of carbon dioxide, blunting of arousal responses with decreased cardiac responses to auditory stimulation, diaphragmatic splinting or fatigue, lowering of vasomotor tone with tachycardia, nasopharyngeal bacterial overgrowth, overheating, alteration of sleep patterns, compromise of cerebral blood flow and upper airway obstruction from distortion of nasal cartilages. Recent studies have, however, shown a significant reduction in substance P in the inferior portion of the olivo-cerebellar complex in SIDS infants which is crucial for the integration of motor and sensory information for the control of head and neck movement. This deficit may explain why some infants are not able to move their faces away from potentially dangerous sleeping environments.

Sleep biosignature of Type 2 diabetes: a case-control study.

AIM: To determine whether or not the sleep disturbances associated with Type 2 diabetes affect the structure of sleep. METHODS: We designed a case-control study in 76 patients with Type 2 diabetes and 76 control subjects without Type 2 diabetes, matched by age, gender, BMI and waist and neck circumferences. A subgroup of 32 patients with Type 2 diabetes was also matched with 64 control subjects without Type 2 diabetes according to apnoea-hypopnoea index score. Examination included an overnight full polysomnography. RESULTS: No differences in the percentage of time spent in either rapid eye movement or non-rapid eye movement sleep were observed between groups; however, patients with Type 2 diabetes had more microarousal events during sleep than control subjects [41.4 (total range 4.0-104.4) vs 20.7 (total range 1.3-94.5) events/h; P < 0.001]. These differences were mainly observed during the non-rapid eye movement sleep [7.4 (total range 0-107.2) vs 0.2 (total range 0-65.2) events/h; P < 0.001]. In addition, sleep variables related to oxygen saturation measures, such as the percentage of time spent with oxygen saturation ≤90%, were significantly greater during the rapid eye movement sleep in patients with Type 2 diabetes [20.3 (total range 0-99.2) vs. 10.5 (total range 0-94.0)%; P = 0.047]. This pattern was maintained in the subgroup of patients matched by apnoea-hypopnaea index. Finally, stepwise regression analyses showed that apnoea-hypopnoea index, the presence of Type 2 diabetes and fasting plasma glucose value were independently associated with the number of microarousals (R2 =0.667). CONCLUSIONS: Type 2 diabetes is associated with an altered sleep structure, with different effects according to rapid eye movement (increase in nocturnal hypoxia) or non-rapid eye movement (increase in sleep fragmentation) sleep.

Parasomnia Overlap Disorder with Sexual Behaviors during Sleep in a Patient with Obstructive Sleep Apnea.

ABSTRACT: Sleep-related abnormal sexual behaviors (sexsomnia) are classified as a subtype of NREM sleep parasomnias. Sexsomnia has been reported as part of parasomnia overlap disorder (POD) in two other patients. We present the case of a 42-year-old male patient with video-polysomnography (vPSG) documented POD. The patient had sleepwalking, sleep-related eating, confusional arousals, sexsomnia, sleeptalking, and REM sleep behavior disorder (RBD). Confusional arousals and RBD were documented during the vPSG. This case had the added complexity of obstructive sleep apnea (OSA) playing a role in sleepwalking and sleep related eating, with good response to nasal continuous positive airway pressure (nCPAP). The sexsomnia did not respond to nCPAP but responded substantially to bedtime clonazepam therapy.

Insomnia with physiological hyperarousal is associated with hypertension.

Previous studies have suggested that insomnia with objective short sleep duration is associated with a higher risk of hypertension, and it has been speculated that the underlying mechanism is physiological hyperarousal. In this study, we tested whether insomnia with physiological hyperarousal measured by Multiple Sleep Latency Test (MSLT), a standard test of sleepiness/alertness, is associated with increased risk of hypertension. Two hundred nineteen chronic insomniacs and 96 normal sleepers were included in this study. Chronic insomnia was defined based on standard diagnostic criteria with symptoms lasting ≥6 months. All subjects underwent 1 night in laboratory polysomnography followed by a standard MSLT. We used the median mean MSLT value (ie, >14 minutes) and the 75th percentile of mean MSLT value (ie, >17 minutes) to define hyperarousal. Hypertension was defined based either on blood pressure measures or on diagnosis treatment by a physician. After controlling for age, sex, body mass index, apnea-hypopnea index, diabetes mellitus, smoking, alcohol, and caffeine use, insomnia combined with MSLT >14 minutes increased the odds of hypertension by 300% (odds ratio=3.27; 95% confidence interval=1.20-8.96), whereas insomnia combined with MSLT >17 minutes increased even further the odds of hypertension by 400% (odds ratio=4.33; 95% confidence interval=1.48-12.68) compared with normal sleepers with MSLT ≤14 minutes. Insomnia associated with physiological hyperarousal is associated with a significant risk of hypertension. Long MSLT values may be a reliable index of the physiological hyperarousal and biological severity of chronic insomnia.

Nocturnal enuresis: an approach to assessment and treatment.

On the basis of strong evidence, although primary monosymptomatic nocturnal enuresis (PMNE) is common and most children will outgrow the condition spontaneously, the psychological effect to the child can be significant and represents the main reason for treatment of these children. On the basis of international consensus panels, treatment of PMNE should be targeted toward the specific type of bedwetting patterns the child has, using bladder diary, sleep history, and daytime elimination concerns as a guide (Table 3). On the basis of international consensus panels, it is important for the primary care physician to be able to differentiate children with PMNE from children with nonmonosymptomatic nocturnal enuresis (NMNE) and secondary nocturnal enuresis. On the basis of international consensus panels, children with NMNE should have their underlying voiding or stool problem addressed before initiation of therapy for the nocturnal enuresis. On the basis of strong evidence, both the bedwetting alarm and desmopressin are considered first-line therapy for children with PMNE.

Mechanisms of sudden unexpected death in epilepsy: the pathway to prevention.

Sudden unexpected death in epilepsy (SUDEP) is the leading cause of death in patients with refractory epilepsy, with an estimated 35% lifetime risk in this patient population. There is a surprising lack of awareness among patients and physicians of this increased risk of sudden death: in a recent survey, only 33% of Canadian paediatricians who treated patients with epilepsy knew the term SUDEP. Controversy prevails over whether cardiac arrhythmia or respiratory arrest is more important as the primary cause of death. Effective preventive strategies in high-risk patients will rely on definition of the mechanisms that lead from seizures to death. Here, we summarize evidence for the mechanisms that cause cardiac, respiratory and arousal abnormalities during the ictal and postictal period. We highlight potential cellular mechanisms underlying these abnormalities, such as a defect in the serotonergic system, ictal adenosine release, and changes in autonomic output. We discuss genetic mutations that cause Dravet and long QT syndromes, both of which are linked with increased risk of sudden death. We then highlight possible preventive interventions that are likely to decrease SUDEP incidence, including respiratory monitoring in epilepsy monitoring units and overnight supervision. Finally, we discuss treatments, such as selective serotonin reuptake inhibitors, that might be personalized to a specific genetic or pathological defect.

[Confusional arousal: a rare cause of self-injurious behaviour]. / Confusional arousal als zeldzame oorzaak van zelfverwonding.

Non-rapid eye movement (NREM) arousal sleep disorders (confusional arousal, somnambulism and sleep terror) are self-limiting and temporary phenomena which cannot be attributed to medical or psychiatric factors. However, very occasionally they can be the cause of unintentional injury to self or others. We describe the case of an 18-year-old who engaged in self-injurious behaviour while asleep. This behaviour could be attributed to confusional arousal.

[Primary monosymptomatic enuresis: diagnostics and therapy]. / Primäre Monosymptomatische Enuresis: Diagnostik und Therapie.

Nocturnal enuresis is one of the most common problems in childhood. In this article a standardized terminology for basic diagnostics additionally to extended diagnostics will be presented. Depending on the findings a specialized therapy can be performed. Besides drug therapy with antidiuretic hormone (ADH) sleep arousal devices can be used and the combination of both approaches also shows excellent results. At the end of therapy a protracted withdrawal shows better results than abrupt cessation.

Nocturnal enuresis.

Nocturnal enuresis (NE) is increasingly seen as part of a heterogeneous phenomenon that at times will include daytime lower urinary tract symptoms such as urgency, frequency and wetting - with reduced bladder storage, usually due to an overactive bladder. In turn, these may be associated with constipation and/or faecal soiling. This paper discusses these considerations in the management of NE.

Decreased spontaneous arousability in preterm newborns with impaired neurological outcome.

Preterm newborns are at high risk of neurological injury. In this population, we investigated the link between neurological complications and sleep architecture. At term-corrected gestational age, we studied retrospectively the polysomnography of 45 preterm infants born at < 28 weeks or weighting < 1 kg. These infants were followed-up by a neuropaediatrician (median age at last follow-up 50.4 months). Two groups of children were constituted: a group without neurological disorder and a second group with at least one of the following: cerebral palsy, language or mental retardation, visual or hearing disability or attention disorder. A Multiple Indicators and Multiple Causes model assessed the relationship between the neurological outcome and two sleep components: spontaneous arousability [number of awakenings and movements per hour of quiet sleep (QS) and active sleep] and QS characteristics (median duration of QS cycles and percentage of QS over total sleep time). Twenty-six infants had an impaired neurological outcome. There were no statistical differences between the two groups regarding clinical characteristics. Compared to preterm neonates with normal neurological outcome, those with impaired outcomes had a lower spontaneous arousability; i.e. 0.7 (0.5­1) times less awakenings and movements per hour of QS and 0.9 (0.8­1) times less per hour of active sleep than infants with normal outcomes (P = 0.05). The differences in QS characteristics did not reach statistical significance. These findings suggested that, in preterm infants, perinatal neurological injuries could be associated with an abnormal sleep architecture characterized by altered spontaneous arousability.

Sleep bruxism, an autonomic self-regulating response by triggering the trigeminal cardiac reflex.

Sleep bruxism, an intensified manifestation of rhythmic masticatory muscle activity, characterized by tooth grinding or clenching during sleep, lacks a definitive physiological purpose. This paper posits that physiologically, sleep bruxism is an autonomic self-regulatory response to nighttime occurrences of tachycardia stemming from the brain experiencing microarousals during sleep. Sleep bruxism by triggering the trigeminal cardiac reflex leads to bradycardia. Rhythmic masticatory muscle activity-sleep bruxism, thereby, serves to slow the heart rate when brain microarousals cause tachycardia.

Nonrestorative sleep as a distinct component of insomnia.

STUDY OBJECTIVES: Explore characteristics of nonrestorative sleep (NRS) in prospectively defined subgroups of individuals with NRS symptoms, investigate whether NRS can occur independently of difficulties initiating and maintaining sleep (DIS/DMS), and determine its effect on waking function. DESIGN: Cross-sectional and longitudinal population-based study comparing patterns of daytime symptoms, and their persistence, in cohorts of subjects with NRS symptoms grouped according to presence or absence of DIS and DMS. SETTING: 28 sleep centers in the US. PARTICIPANTS: Subjects reporting awakening unrestored or unrefreshed at least 3 times weekly over the previous 3 months were classified, based on self-reported sleep problems, to DIS (n = 138), DMS (n = 44), DIS+DMS (n = 125), and NRS-only (no DIS or DMS; n = 192) cohorts. Eighty healthy volunteers formed a control group. INTERVENTIONS: None. MEASUREMENTS AND RESULTS: Polysomnography confirmed DIS and/or DMS in 56/138 (41%), 18/44 (41%), and 37/125 (30%) subjects in DIS, DMS, and DIS+DMS cohorts, respectively; and absence of DIS or DMS in 115/192 (60%) NRS-only subjects and 52/80 (65%) healthy volunteers. Multiple subject-reported endpoints including the Endicott Work Productivity Scale, Pittsburgh Insomnia Rating Scale, Restorative Sleep Questionnaire, and SF-36, showed that NRS-only subjects had significantly impaired daytime function relative to healthy volunteers, comparable to impairment affecting subjects with DIS and/or DMS. Symptoms persisted over 3 months. CONCLUSIONS: This study confirms that NRS can occur independently of other components of insomnia. Daytime symptoms were as severe in individuals with NRS-only as those whose NRS symptoms were combined with DIS or DMS.

Sleep and arousal as risk factors for adverse health and work performance in public health workers involved in the 2004 Florida hurricane season.

BACKGROUND: We examined the relation of sleep disturbance and arousal to work performance, mental and physical health, and day-to-day functioning in Florida Department of Health (FDOH) employees 9 months after the 2004 Florida hurricane season. METHODS: FDOH employees were contacted via e-mail 9 months after the 2004 hurricanes. Participants (N = 2249) completed electronic questionnaires including measures of sleep disturbance, arousal, work performance, physical health, mental health, day-to-day function, hurricane injury, and work demand. RESULTS: More than 18% of FDOH employees reported ≥ 25% reduced work performance and 11% to 15.3% reported ≥ 7 "bad" mental or physical health days in the past month. Sleep disturbance and elevated arousal were strongly associated with impaired work performance (odds ratios [ORs] 3.33 and 3.34, respectively), "bad" mental health (ORs 3.01 and 3.64), "bad" physical health (ORs 3.21 and 2.01), and limited day-to-day function (ORs 4.71 and 2.32), even after adjusting for sex, race, age, education, and marital status. CONCLUSIONS: Among public health workers exposed to the 2004 hurricanes, sleep disturbance and arousal were associated with personal and work impairment. Future research should continue to examine the effect of repeated exposure to disasters in first responders.

Nocturnal polyuria and nocturnal arginine vasopressin (AVP): a key factor in the pathophysiology of monosymptomatic nocturnal enuresis.

AIMS: To identify the relationship between nocturnal AVP deficiency, nocturnal polyuria (NP), and low urinary osmolality in children suffering of primary monosymptomatic nocturnal enuresis (NE). PATIENTS AND METHODS: The study included 50 children (28 males and 22 females) with primary monosymptomatic NE and 30 non enuretic children of the same age group (controls). Night samples of blood and urine were obtained for AVP, blood osmolality, and urine osmolality. In addition, volume frequency charts, arousal threshold, and urodynamics were performed for these children. RESULTS: Twenty eight (56%) of the enuretic children were considered to have NP. Mean AVP level was 44.80 +/- 8.19 and 32.49 +/- 18.25 pg/ml while mean urine osmolality was 865.07 +/- 158.66 mOsm/kg and 700.06 +/- 84.42 mOsm/kg in controls and enuretic group respectively. These differences were highly significant. No significant difference was found between the controls and enuretics without NP. On the other hand, nocturnal AVP and urine osmolality were significantly lower in enuretics with NP when compared to both controls and enuretics without NP. Blood osmolality did not reach statistically significant difference between subgroups. Arousal threshold was significantly higher in enuretic children irrespective to NP. The timing for NE episodes were predominantly late in the night in NE children without NP while patients suffering of NE with NP typically experienced multiple incidents each night. CONCLUSION: We have shown that low nocturnal AVP and urine osmolality may play a role in the pathophysiology of enuretics with NP. This abnormality doesn't occur as an isolated disease as these children suffer from arousal defect as well.