Min Yu, Zeliang Hao, Liyue Xu, Long Zhao, Yongfei Wen, Fang Han, Xuemei Gao
{"title":"Catathrenia 表型的多导睡眠图和颅面特征的差异:聚类分析","authors":"Min Yu, Zeliang Hao, Liyue Xu, Long Zhao, Yongfei Wen, Fang Han, Xuemei Gao","doi":"10.2147/nss.s455705","DOIUrl":null,"url":null,"abstract":"<strong>Purpose:</strong> 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.<br/><strong>Patients and Methods:</strong> 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.<br/><strong>Results:</strong> 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, <em>p</em>=0.005), and broader upper airway (volume 41,386± 10,543 and 26,661± 6700 mm<sup>3</sup>, <em>p</em>< 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, <em>p</em>=0.009), more respiratory effort-related arousals (RERA)(1 [1, 109] and 32 [13, 57)], <em>p</em>=0.005), smaller upper airway (cross-sectional area of velopharynx 512± 87 and 339± 84 mm<sup>2</sup>, <em>p</em>< 0.001) and better response to treatment (41.2% and 82.6%, <em>p</em>=0.004).<br/><strong>Conclusion:</strong> 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.<br/><br/><strong>Keywords:</strong> subtype, groaning, upper airway, treatment, OSA, sleep-disordered breathing<br/>","PeriodicalId":18896,"journal":{"name":"Nature and Science of Sleep","volume":null,"pages":null},"PeriodicalIF":3.0000,"publicationDate":"2024-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Differences in Polysomnographic and Craniofacial Characteristics of Catathrenia Phenotypes: A Cluster Analysis\",\"authors\":\"Min Yu, Zeliang Hao, Liyue Xu, Long Zhao, Yongfei Wen, Fang Han, Xuemei Gao\",\"doi\":\"10.2147/nss.s455705\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<strong>Purpose:</strong> 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.<br/><strong>Patients and Methods:</strong> 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.<br/><strong>Results:</strong> 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, <em>p</em>=0.005), and broader upper airway (volume 41,386± 10,543 and 26,661± 6700 mm<sup>3</sup>, <em>p</em>< 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, <em>p</em>=0.009), more respiratory effort-related arousals (RERA)(1 [1, 109] and 32 [13, 57)], <em>p</em>=0.005), smaller upper airway (cross-sectional area of velopharynx 512± 87 and 339± 84 mm<sup>2</sup>, <em>p</em>< 0.001) and better response to treatment (41.2% and 82.6%, <em>p</em>=0.004).<br/><strong>Conclusion:</strong> 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.<br/><br/><strong>Keywords:</strong> subtype, groaning, upper airway, treatment, OSA, sleep-disordered breathing<br/>\",\"PeriodicalId\":18896,\"journal\":{\"name\":\"Nature and Science of Sleep\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":3.0000,\"publicationDate\":\"2024-05-29\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Nature and Science of Sleep\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.2147/nss.s455705\",\"RegionNum\":2,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"CLINICAL NEUROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Nature and Science of Sleep","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.2147/nss.s455705","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
Differences in Polysomnographic and Craniofacial Characteristics of Catathrenia Phenotypes: A Cluster Analysis
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.
Keywords: subtype, groaning, upper airway, treatment, OSA, sleep-disordered breathing
期刊介绍:
Nature and Science of Sleep is an international, peer-reviewed, open access journal covering all aspects of sleep science and sleep medicine, including the neurophysiology and functions of sleep, the genetics of sleep, sleep and society, biological rhythms, dreaming, sleep disorders and therapy, and strategies to optimize healthy sleep.
Specific topics covered in the journal include:
The functions of sleep in humans and other animals
Physiological and neurophysiological changes with sleep
The genetics of sleep and sleep differences
The neurotransmitters, receptors and pathways involved in controlling both sleep and wakefulness
Behavioral and pharmacological interventions aimed at improving sleep, and improving wakefulness
Sleep changes with development and with age
Sleep and reproduction (e.g., changes across the menstrual cycle, with pregnancy and menopause)
The science and nature of dreams
Sleep disorders
Impact of sleep and sleep disorders on health, daytime function and quality of life
Sleep problems secondary to clinical disorders
Interaction of society with sleep (e.g., consequences of shift work, occupational health, public health)
The microbiome and sleep
Chronotherapy
Impact of circadian rhythms on sleep, physiology, cognition and health
Mechanisms controlling circadian rhythms, centrally and peripherally
Impact of circadian rhythm disruptions (including night shift work, jet lag and social jet lag) on sleep, physiology, cognition and health
Behavioral and pharmacological interventions aimed at reducing adverse effects of circadian-related sleep disruption
Assessment of technologies and biomarkers for measuring sleep and/or circadian rhythms
Epigenetic markers of sleep or circadian disruption.