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Purpose: Catathrenia is a rare sleeping disorder characterized by repetitive nocturnal groaning during prolonged expirations. Patients with catathrenia had heterogeneous polysomnographic, comorbidity, craniofacial characteristics, and responses to treatment. Identifying phenotypes of catathrenia might benefit the exploration of etiology and personalized therapy. Patients and Methods: Sixty-six patients diagnosed with catathrenia by full-night audio/video polysomnography seeking treatment with mandibular advancement devices (MAD) or continuous positive airway pressure (CPAP) were included in the cohort. Polysomnographic characteristics including sleep architecture, respiratory, groaning, and arousal events were analyzed. Three-dimensional (3D) and 2D craniofacial hard tissue and upper airway structures were evaluated with cone-beam computed tomography and lateral cephalometry. Phenotypes of catathrenia were identified by K-mean cluster analysis, and inter-group comparisons were assessed. Results: Two distinct clusters of catathrenia were identified: cluster 1 (n=17) was characterized to have more males (71%), a longer average duration of groaning events (18.5±4.8 and 12.8±5.7s, p=0.005), and broader upper airway (volume 41,386±10,543 and 26,661±6700 mm3, p<0.001); cluster 2 (n=49) was characterized to have more females (73%), higher respiratory disturbance index (RDI) (median 1.0 [0.3, 2.0] and 5.2 [1.2, 13.3]/h, p=0.009), more respiratory effort-related arousals (RERA)(1 [1, 109] and 32 [13, 57)], p=0.005), smaller upper airway (cross-sectional area of velopharynx 512±87 and 339±84 mm2, p<0.001) and better response to treatment (41.2% and 82.6%, p=0.004). Conclusion: Two distinct phenotypes were identified in patients with catathrenia, primary catathrenia, and catathrenia associated with upper airway obstruction, suggesting respiratory events and upper airway structures might be related to the etiology of catathrenia, with implications for its treatment.

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STUDY OBJECTIVES: The International Classification of Sleep Disorders categorized catathrenia as a respiratory disorder, but there are doubts whether episodes appear during rapid eye movement (REM) sleep or the non-rapid eye movement (NREM), their duration, and symptoms. The main objectives were to identify the most common features and relations of catathrenia. METHODS: PubMed, Embase, and Web of Science were searched according to the PRISMA 2020 guidelines. The Joanna Briggs Institute and the ROBINS-I tools were chosen to assess the risk of bias. RESULTS: A total of 288 records were identified, 31 articles were included. The majority of the studies had a moderate risk of bias. 49.57% of episodes occurred during the NREM sleep, while 46% took place during REM. In 60.34% females, catathrenia was more common in the NREM, while in 59.26% of males was in REM sleep (p < 0.05). Females and obese individuals were found to have shorter episodes (p < 0.05). Age was inversely correlated with minimal episodes duration (r = - 0.34). The continuous positive airway pressure (CPAP) therapy was inversely correlated with the maximal episode duration (r = - 0.48). CONCLUSIONS: Catathrenia occurs with similar frequency in both genders. The most frequent symptoms embraced groaning, awareness of disturbing bedpartners, and daytime somnolence-not confirmed by the Epworth Sleepiness Scale. The episodes occur more frequently in NREM than in REM sleep. Catathrenia may be considered as a sex-specific condition. The effects of CPAP treatment leading to shortening episodes duration, which may indicate the respiratory origin of catathrenia.

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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.

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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.

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STUDY OBJECTIVES: To determine the clinical presentation, polysomnographic appearance, and the response to therapy of catathrenia in children. METHODS: A retrospective analysis was performed in children referred or evaluated at the Boston Children's Hospital Sleep Center between 1998 and 2021 who were documented to have catathrenia either as a chief complaint or an incidental finding. RESULTS: Catathrenia was identified in 21 children. The age of onset by parent report was 6.4 ± 4.4 years (range: birth-14 years), but the diagnosis was at 8.3 ± 3.8 years (range: 1.5-14 years). Catathrenia was the chief complaint for 24% of patients and was incidentally identified in 76% referred for breathing concerns. Bruxism was observed in 62% of patients. Catathrenia events most often occur following an electrocortical arousal (79%) and are most common in the second half of the night (73%). Catathrenia was observed exclusively in rapid eye movement (REM) sleep (16%), exclusively in non-REM sleep (32%), and in both sleep states (52%). Treatment of obstructive sleep apnea resulted in resolution or improvement in the obstructive sleep apnea in all patients, but the catathrenia persisted at a reduced level. CONCLUSIONS: Most catathrenia in children is diagnosed incidentally during evaluation for sleep-disordered breathing, although the groaning often started many years earlier. Catathrenia events decreased considerably after treatment of obstructive sleep apnea, but persisted in all patients. Catathrenia was also observed in children without signs or symptoms of sleep-disordered breathing, indicating that these are distinct conditions. The majority of children with catathrenia had no sleepiness or behavioral concerns. CITATION: Katz ES, D'Ambrosio C. Catathrenia in children: clinical presentation, polysomnographic features, natural history, and response to therapy. J Clin Sleep Med. 2023;19(8):1505-1511.

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Background: Purposeless groaning is primarily encountered in patients with progressive supranuclear palsy and has also been reported to occur in advanced Parkinson's disease (PD). Objective: We describe a case of pronounced purposeless groaning occurring as a medication OFF-period nonmotor phenomenon in PD. To our knowledge, this has not been previously reported in the literature. Methods and Materials: We describe and provide video documentation of a patient with moderately advanced PD and motor fluctuations, in whom OFF-period groaning was reported by the family and observed during clinic consultations to be a prominent feature, occupying approximately 40% of his OFF periods as calculated from his PD diary. Conclusions: Although rare, OFF-period purposeless groaning in PD can be very disruptive and add significantly to caregiver burden. It is postulated to be a disinhibitory and perseverative behavior related to overactivation of the cingulo-periaqueductal circuit; further study is needed to delineate the underlying pathophysiological mechanisms.

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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.

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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.

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Involuntary moaning has been reported in sporadic cases of neurodegenerative diseases. A five-generation Hispanic family with eight members exhibiting involuntary moaning, most of whom with isolated moaning in the absence of any additional neurological disorder carried a missense variant in the NEFH gene segregating in the family.

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STUDY OBJECTIVES: Catathrenia is a rare disease, classified as isolated symptoms and normal variants under sleep-related breathing disorders in the International Classification of Sleep Disorders, third edition. Because of its rarity, the research on its pathogenesis and treatment is insufficient. This study aimed to evaluate whether the mandibular advancement device (MAD) could be considered an alternative treatment trial and if so, to explore factors predicting its effectiveness. METHODS: Thirty patients (12 men and 18 women, aged 16-67 years) with catathrenia participated in the study. They underwent standard clinical evaluation, questionnaires, physical examinations, craniofacial evaluations, video polysomnography, and imaging of the upper airway before and after the insertion of the MAD. The groaning index (groaning episodes per hour of sleep) and apnea-hypopnea index were evaluated, and anatomic factors predicting effectiveness were explored. RESULTS: The sleep efficiency of most patients was higher than 80%, and groaning was present throughout all stages of sleep. With the insertion of the MAD, the groaning index decreased significantly from 5.8 (2.7-14.3) to 2.8 (1.3-12.2) events/h (P = .014). Age had a negative effect on efficacy. Mandibular repositioning of the MAD, especially the amount of vertical opening and changes of the cross-sectional area of the hypopharynx, was positively related with efficacy. CONCLUSIONS: The MAD could be considered a possible treatment trial for those seeking treatment for groaning. CLINICAL TRIAL REGISTRATION: Registry: Chinese Clinical Trial Register; Name: The etiology of catathrenia based on oral and maxillofacial system; URL: http://www.chictr.org.cn/showproj.aspx?proj=22286; Identifier: ChiCTR-COC-17013239. CITATION: Yu M, Hao Z, Xu L, Wen Y, Han F, Gao X. Mandibular advancement device as treatment trial for catathrenia (nocturnal groaning). J Clin Sleep Med. 2021;17(9):1767-1776.

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Background: Immunological causes of atypical parkinsonisms linked to neuronal specific antibodies have been recently reported. As these are potentially treatable disorders, it is desirable to identify which clinical features may suggest an autoimmune etiology. Case Report: A 60-year-old-man with progressive supranuclear palsy associated with anti-LGI-1 antibodies presented with rapidly progressive dementia and moaning. Treatment with steroids and immunoglobulin resulted in temporary clinical improvement and disease stabilization. Discussion: Anti-LGI-1 antibodies interfere with normal synaptic activity and maturation in the central nervous system. We suggest that an immune-mediated mechanism might be considered in atypical parkinsonisms with unusual features such as rapidly progressive dementia. Highlights: We present a case of rapidly evolving progressive supranuclear palsy-like parkinsonism associated with anti-LGI-1 antibodies, suggesting that immune-mediated mechanisms might be involved in rapid progression of some atypical parkinsonisms. This case also contributes to the expanding spectrum of moaning-associated disorders.

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Purposeless groaning has been reported in advanced progressive supranuclear palsy. We present a case of purposeless groaning occurring as a primary complaint in a patient with advanced Parkinson's disease. Purposeless groaning is thought to be a manifestation of disinhibition and perseveration due to frontal-subcortical dysfunction. Proper recognition of this phenomenon will help clinicians to avoid unnecessary investigations and treatment (e.g., prescription of opioid medications).

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Purposeless groaning has been reported in advanced progressive supranuclear palsy. We present a case of purposeless groaning occurring as a primary complaint in a patient with advanced Parkinson's disease. Purposeless groaning is thought to be a manifestation of disinhibition and perseveration due to frontal-subcortical dysfunction. Proper recognition of this phenomenon will help clinicians to avoid unnecessary investigations and treatment (e.g., prescription of opioid medications).

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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.

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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.

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