RESUMEN

Parasomnias and sleep-related movement disorders (SRMD) are major causes of sleep disorders and may be drug induced. The objective of this study was to conduct a systematic review of the literature to examine the association between drug use and the occurrence of parasomnias and SRMD. Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines for reporting systematic reviews, we searched PubMed databases between January 2020 and June 2023. The searches retrieved 937 records, of which 174 publications were selected for full-text screening and 73 drugs were identified. The most common drug-induced parasomnias were nightmares and rapid eye movement (REM) sleep behaviour disorders and sleepwalking. In terms of drug-induced SRMD, restless legs syndrome, periodic limb movement disorders (PLMD), and sleep-related bruxism were most frequent. Medications that inhibit noradrenergic, serotonergic, or orexin transmission could induce REM sleep (e.g., nightmares). Regarding sleepwalking, dysregulation of serotoninergic neurone activity is implicated. Antipsychotics are mentioned, as well as medications involved in the gamma-aminobutyric acid (GABA) pathway. A mechanism of desensitisation-autoregulation of GABA receptors on serotoninergic neurones is a hypothesis. SRMD and PLMD could involve medications disrupting the dopamine pathway (e.g., antipsychotics or opioids). Opioids would act on mu receptors and increase dopamine release. The role of adenosine and iron is also hypothesised. Regarding bruxism, the hypotheses raised involve dysregulation of mesocortical pathway or a downregulation of nigrostriatal pathway, related to medications involving dopamine or serotonin. Parasomnias are rarely identified in drug product labels, likely due to the recent classification of their diagnoses. An analysis of pharmacovigilance data could be valuable to supplement existing literature data.

RESUMEN

Catathrenia is a loud expiratory moan during sleep that is a social embarrassment and is sometimes confused with central apnea on polysomnography. It affects about 4% of adults, but cases are rarely referred to sleep centers. Catathrenia affects males and females, children and adults, who are usually young and thin. A "typical" catathrenia begins with a deep inhalation, followed by a long, noisy exhalation, then a short, more pronounced exhalation, followed by another deep inhalation, often accompanied by arousal. The many harmonics of the sound indicate that it is produced by the vocal cords. It is often repeated in clusters, especially during REM sleep and at the end of the night. It does not disturb the sleepers, but their neighbors, and is associated with excessive daytime sleepiness in one-third of cases. The pathophysiology and treatment of typical catathrenia are still unknown. Later, a more atypical catathrenia was described, consisting of episodes of short (2 s), regular, semi-continuous expiratory moans during NREM sleep (mainly in stages N1 and N2) and REM sleep, often in people with mild upper airway obstruction. This atypical catathrenia is more commonly reduced by positive airway pressure and mandibular advancement devices that promote vertical opening.

Asunto(s)

RESUMEN

STUDY OBJECTIVES: Catathrenia, derived from the Greek κατά (kata) meaning below and θρηνώ (threnia) to lament, is characterized by expiratory groaning episodes during sleep. In a case series of nine patients with severe obstructive sleep apnea, we observed a peculiar groaning entity that has not been described before. METHODS: We described and illustrated the cases with polysomnographic tracings and additional audio recordings. RESULTS: All patients were men, obese (body mass index 39 ± 6 kg/m2) with an apnea-hypopnea index ranging from 47 to 125/h. In addition, we identified groaning events that were consistently preceded by a cortical arousal associated with a "rescue" respiration after an obstructive hypopnea or apnea. These events exhibited characteristics of "mixed apnea's", but the "central apnea-like part" was a prolonged expiratory groaning phase, with immediately after the terminal expiratory snort appearance of an obstructive apnea. In case the duration of this expiration was at least 10 s we calculated these events separately and the index was 8.4 ± 7.7/h. More rarely (index 0.6 ± 0.5/h) a "central apnea mimicking event" with groaning not followed by an obstruction, was observed. We also observed groaning episodes during expiration with a shorter duration (less than 10 s), not calculated separately. Positive airway pressure, which was well tolerated, eliminated these events. CONCLUSIONS: This novel catathrenia entity preceded by a cortical arousal and "rescue" respiration in response to obstructive events is intriguing. Possible explanations for these observations are further discussed in this article.

Asunto(s)

RESUMEN

STUDY OBJECTIVES: Fatal insomnia (FI) is a rare prion disease severely affecting sleep architecture. Breathing during sleep has not been systematically assessed. Our aim was to characterize the sleep architecture, respiratory patterns, and neuropathologic findings in FI. METHODS: Eleven consecutive FI patients (ten familial, one sporadic) were examined with video-polysomnography (vPSG) between 2002 and 2017. Wake/sleep stages and respiration were evaluated using a modified scoring system. Postmortem neuropathology was assessed in seven patients. RESULTS: Median age at onset was 48 years and survival after vPSG was 1 year. All patients had different combinations of breathing disturbances including increased respiratory rate variability (RRV; n = 7), stridor (n = 9), central sleep apnea (CSA) (n = 5), hiccup (n = 6), catathrenia (n = 7), and other expiratory sounds (n = 10). RRV in NREM sleep correlated with ambiguous and solitary nuclei degeneration (r = 0.9, p = 0.008) and reduced survival (r = -0.7, p = 0.037). Two new stages, Subwake1 and Subwake2, present in all patients, were characterized. NREM sleep (conventional or undifferentiated) was identifiable in ten patients but reduced in duration in eight. REM sleep occurred in short segments in nine patients, and their reduced duration correlated with medullary raphe nuclei degeneration (r = -0.9, p = 0.005). Seven patients had REM without atonia. Three vPSG patterns were identified: agitated, with aperiodic, manipulative, and finalistic movements (n = 4); quiet-apneic, with CSA (n = 4); and quiet-non-apneic (n = 3). CONCLUSIONS: FI patients show frequent breathing alterations, associated with respiratory nuclei damage, and, in addition to NREM sleep distortion, have severe impairment of REM sleep, related with raphe nuclei degeneration. Brainstem impairment is crucial in FI.

Asunto(s)

RESUMEN

Two patients diagnosed with Nocturnal Groaning were treated with intramuscular injection of botulinum toxin type A (BoNT/A) in the thyroarytenoid muscle with significant reduction of groaning episodes. Treatment with BoNT/A may be an effective therapy of Nocturnal Groaning, but large clinical studies are needed to determine its role in this clinical setting.

Asunto(s)

RESUMEN

Catatrenia (gemido nocturno) es una condición rara caracterizada por sonidos irregulares que ocurren durante el sueño. Los comportamientos ocurren intermitentemente durante cualquiera de las dos etapas de sueño, REM o NREM y se caracterizan por gemidos prolongados, a menudo muy fuertes, socialmente perturbadores, durante la expiración. Es poco conocido y espera más definición y estudios terapéuticos. Hay pocos reportes y en su mayoría de pacientes adultos. Se presentan 3 casos en pacientes pediátricos.

Catathrenia (nocturnal groaning) is a rare condition characterized by irregular sounds that occur during sleep. The behaviors occur intermittently during either REM or NREM sleep and are characterized by prolonged, often very loud, socially disruptive groaning sounds during expiration. It is poorly understood and awaits further definition and therapeutic studies. There are few reports mostly adult patients are presented below 3 cases in pediatric patients.

Asunto(s)

RESUMEN

NONE: During upward titration of a dose of sodium oxybate therapy for narcolepsy with cataplexy, a 25-year-old woman was observed by her husband to have new onset of knuckle-cracking and moaning behaviors during sleep ≥1 nights each week. The patient did previously occasionally crack her knuckles during the day (but never at night). These behaviors had not been evaluated by polysomnography. After transition of care, polysomnography with video monitoring was ordered and revealed 2 knuckle-cracking episodes that developed out of stage N2 sleep and were likely a non-rapid eye movement sleep parasomnia associated with sodium oxybate treatment.

Asunto(s)

RESUMEN

Two young males with refractory epilepsy of unknown aetiology were referred for vagus nerve stimulation (VNS). Sleep disturbances emerged following VNS parameter changes. In Patient 1, video-polysomnogram (PSG) disclosed snoring and catathrenia in non-REM sleep. Central apnoea also occurred, but more rarely. In Patient 2, video-PSG showed mixed apnoea with desaturation and episodes of stridor followed by a catathrenia-like sound. A drug-induced sleep endoscopy (DISE) revealed, during VNS OFF time, glossoptosis, "trap door" of the epiglottis, and paresis of the left side of the larynx and ipsilateral vocal cords. During ON time, there were periods of pharyngeal collapse, in which video-PSG revealed patterns suggestive of both obstructive and central sleep apnoea. All these sleep-related phenomena were coincident with VNS ON time. In the first patient, VNS parameter adjustment was sufficient to successfully reverse all the symptoms, whereas the other patient required concomitant treatment with continuous positive airway pressure. The data broaden our knowledge about sleep disorders related to VNS, in particular stridor and catathrenia. We suggest that central sleep apnoea may be associated with laryngeal occlusion. DISE may be considered in selected cases as a valuable clinical tool to evaluate, in a single session, the effectiveness of multiple VNS parameter changes on respiration and laryngeal side effects. [Published with video sequences].

Asunto(s)

RESUMEN

None: Catathrenia, also known as sleep-related groaning, is a relatively rare sleep disorder with characteristics consistent with loud groaning on expiration during sleep. Organic causes of catathrenia are unknown and the decision of whether or not to treat is unclear. Limited research is available concerning appropriate treatment and none of the literature focuses primarily on pediatrics. We report a series of three, male pediatric cases with catathrenia that were treated with continuous positive airway pressure (CPAP) while identifying and comparing clinical symptoms, polysomnogram findings, treatment, and patient response to treatment. Catathrenia may be associated with abnormal nocturnal oxygenation and ventilation and may lead to negative clinical daytime symptoms which may warrant treatment. If catathrenia leads to sleep disruption and negative daytime symptoms, treatment with CPAP should be considered.

Asunto(s)

RESUMEN

ABSTRACT: Catathrenia is a rare sleep-related breathing disorder marked by nocturnal groaning during prolonged exhalation. Home sleep apnea tests are growing in popularity, but no prior case reports exist documenting catathrenia detected by a home sleep apnea test. We report two cases of catathrenia incidentally detected in patients with low to intermediate risk of obstructive sleep apnea, in which diagnostic polysomnography was recommended but denied by insurers. Events were automatically scored as central apneas, leading to diagnostic uncertainty. The key to identification of catathrenia in both patients was combination of audio review of events, pattern of expiratory air trapping, and mismatch between central apnea flow signal attenuation and increased sound noted on audio amplitude signal. Diagnostic polysomnography was ordered in both cases for confirmation of diagnosis, leading to increased care costs.

Asunto(s)

Asunto(s)

RESUMEN

Catathrenia is a rare sleep disease characterized by monotonous groaning sounds that appear to be related with prolonged expiration, commonly experienced during rapid eye movement (REM) sleep. Catathrenia is also known as nocturnal groaning or sleep-related groaning and is currently categorized as a sleep-related breathing disorder. We present a rare case of a 19-year-old male with nocturnal groaning during non-REM sleep. We suggest that if catathrenia is suspected, polysomnography should be utilized to differentiate it from various sleep disorders such as snoring, central sleep apnea, sleep talking, parasomnia, and sleep-related movement disorders.

Asunto(s)

Asunto(s)

RESUMEN

STUDY OBJECTIVES: Catathrenia is an underrecognized nocturnal vocalization phenomenon that can be a source of perplexity to patients, bed partners, and medical providers. Catathrenia is distinct from both sleep talking (a parasomnia with loud talking during sleep) and snoring (noise due to vibration of upper airway soft tissues related to variations in airway resistance). The objective of this review is to provide an evidence-based resource to help the practitioner reliably evaluate and manage patients with this condition. METHODS: Data were gathered from: (1) PubMed, Scopus, Web of Science, and Google Scholar; and (2) catathrenia social media groups (Yahoo and Facebook). RESULTS: Data collected were (1) 15 case reports and 17 case series describing 191 patients with catathrenia; (2) questionnaires from 47 catathrenia subjects; (3) 5 audio files. CONCLUSIONS: Catathrenia is a noise produced during sleep (distinct from snoring) with identifiable harmonics, a computable main frequency, and high-decibel intensity that involves active adduction and vibration of the vocal cords during expiration. The quality of groaning in catathrenia is monotone, and often presents with a morose or sexual connotation, causing a significant social problem for patients. Although there is no association with risk of physical harm, catathrenia does present a significant disturbance to the bed partner and has been associated with subjective impairments to sleep quality, including unrefreshing sleep and fatigue. Polysomnography can be useful if performed properly to confirm the diagnosis and to evaluate for comorbid sleep disturbances, such as obstructive sleep apnea or parasomnia. Directions for further research could involve consideration of deep breathing exercises, yoga, meditation, or myofunctional therapy to help abate symptoms.

Asunto(s)

RESUMEN

ABSTRACT: Cyclic alternating pattern (CAP) is widely recognized as an expression of sleep instability in electroencephalogram activity during non-rapid eye movement sleep. We report a case with sequences of CAP followed by bruxism and catathrenia in a 10-y-old male patient with a diagnosis of attention deficit hyperactivity disorder in treatment with methylphenidate. We found CAP in 83.1% of all episodes of catathrenia, and the CAP rate was 12.8%. We propose to consider catathrenia as one of the sleep disorders that may be accompanied by CAP.

Asunto(s)

RESUMEN

OBJECTIVES: Catathrenia is an uncommon and poorly understood disorder, characterized by groaning during sleep occurring in tandem with prolonged expiration. Its classification, pathogenesis, and clinical relevance remain debated, substantially due to the limited number of cases reported to date. We report a series of consecutive cases of catathrenia, their clinical and polysomnographic characteristics, and their subsequent management. METHODS: Consecutive patients with catathrenia who had undergone full polysomnography in our institution over a 5.5-year period were included. Catathrenia events (CEs) were examined in clusters, which formulated catathrenia periods (CPs). The relationships between CPs, sleep stage distribution, electroencephalogram (EEG) arousals, and other sleep parameters were assessed, along with the clinical presentation and management of catathrenic patients. RESULTS: A total of 427 CPs were identified in 38 patients, 81% arising from rapid eye movement (REM) sleep. EEG arousals preceded or coincided with the onset of 84% of CPs, which were of longer duration than those not associated with an arousal (57.3 ± 56.8 vs. 32.2 ± 29.4 s, p < 0.001). Each CE had a characteristic airflow signal, with inspiration preceding a protracted expiration and a brief more rapid exhalation, followed by deep inspiration. Although the majority of patients were referred on the basis of bed partner complaints, 44.7% complained of daytime sleepiness. Continuous positive airway pressure therapy and sleep-consolidating pharmacotherapy led to subjective improvement, but were limited by poor long-term adherence. CONCLUSIONS: In the largest series of catathrenia patients reported to date, we found that this rare disorder is characterized by a distinct breathing pattern and arises predominantly from REM sleep, with arousals almost uniformly preceding or coinciding with the onset of CPs.

Asunto(s)

Asunto(s)

RESUMEN

BACKGROUND: Catathrenia is a rare sleep disorder characterized by repeated groaning in a protracted expiration preceded by a deep inspiration. This study aimed to explore whether anatomy is one of pathophysiology of catathrenia by investigating the anatomical features associated with catathrenia in the upper airway, craniofacial structures, and dental patterns. MATERIAL AND METHODS: Twenty-two patients with catathrenia (7 males, 15 females; age 22 to 69 years) were recruited as well as 66 patients matched by age and gender (matching proportion 1:3) with obstructive sleep apnea syndrome (OSAS). Both groups underwent cephalograms and dental casting, and cephalometric measurements and the Peer Assessment Rating (PAR) index was applied. Differences between the two groups were evaluated and cephalometric measurements in catathrenia group were compared with control values of Chinese patients from previous studies. RESULTS: As for airway-related measurements, increased PNS-R, PNS-UPW, and H-FH and decreased SPT and TGL were found in catathrenia group compared to normal values. Such trends were found even more evident when compared with the OSAS Group. As for craniofacial parameters, values of U1/NA and U1/SN were found increased in the catathrenia group compared with normal values and values of MP/FH and Y decreased. The differences were more distinct from the OSAS Group. Increased arch lengths and upper inter-first molar widths, and decreased overbite and PAR index, were found in catathrenia group compared with the OSAS Group. CONCLUSION: Catathrenia patients present with a broad upper airway, yet protrusive upper incisors and flat mandibular angles. Anatomical characteristics of catathrenia are different from those associated with OSAS, namely a wide airway, large skeleton, and good occlusion.

Asunto(s)

RESUMEN

It is a significant challenge for the clinician to make distinction between nocturnal epilepsy form non-epileptic sleep disorders. Although in some patients, diagnosis is easy to achieve but sometimes not. At times even with help of polysomnogram and electroencephalogram, diagnostic confusion remains. We present two cases of nocturnal paroaxysmal events, which still need elucidate diagnosis.

Asunto(s)

RESUMEN

BACKGROUND: The International Classification of Sleep Disorders-Third Edition (ICSD-3) classifies catathrenia among the respiratory disorders and not as a parasomnia as in ICSD-2. Few patients have been reported during these years, and the clinical description of the sound is different from group to group. In fact, there is no full agreement about its nature, origin, meaning, and treatment. METHODS AND RESULTS: In this paper we review the literature on catathrenia focusing on the characteristics of the sound, demographics of the patients, aetiology, response to treatment, etc., in order to support its classification as a respiratory disorder or a parasomnia. We also discuss the possibility of Catathrenia being not one disorder but two variants or two different disorders.

Asunto(s)