Differences in Polysomnographic and Craniofacial Characteristics of Catathrenia Phenotypes: A Cluster Analysis

IF 3 2区医学 Q2 CLINICAL NEUROLOGY

Nature and Science of Sleep Pub Date : 2024-05-29 DOI:10.2147/nss.s455705

Min Yu, Zeliang Hao, Liyue Xu, Long Zhao, Yongfei Wen, Fang Han, Xuemei Gao

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Abstract

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

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Catathrenia 表型的多导睡眠图和颅面特征的差异：聚类分析

目的：夜间呻吟症是一种罕见的睡眠障碍，其特征是在夜间长时间呼气时反复发出呻吟声。catathrenia 患者的多导睡眠图、合并症、颅面特征和对治疗的反应各不相同。确定 catathrenia 的表型可能有助于探索病因和个性化治疗：通过整夜音频/视频多导睡眠图诊断出的66名 catathrenia 患者被纳入队列，他们正在寻求下颌前突装置（MAD）或持续气道正压（CPAP）治疗。对包括睡眠结构、呼吸、呻吟和唤醒事件在内的多导睡眠图特征进行了分析。采用锥束计算机断层扫描和侧向头颅测量法对三维和二维颅面硬组织和上气道结构进行了评估。通过 K-均值聚类分析确定了白内障的表型，并对组间比较进行了评估：结果：发现了两个不同的颅内压增高群：群 1（n=17）的特点是男性较多（71%），呻吟事件的平均持续时间较长（18.5± 4.8 秒和 12.8± 5.7 秒，p=0.005），上气道更宽（容积分别为 41 386±10 543 和 26 661±6700 mm3，p< 0.001）；第 2 组（n=49）的特点是女性较多（73%），呼吸紊乱指数（RDI）较高（中位数为 1.0[0.3，2.0]和5.2[1.2，13.3]/h，p=0.009），更多的呼吸努力相关唤醒（RERA）（1[1，109]和32[13，57]]，p=0.005），更小的上气道（咽部横截面积512± 87和339± 84 mm2，p< 0.001）和更好的治疗反应（41.2%和82.6%，p=0.004）：原发性呻吟和呻吟伴有上气道阻塞的患者有两种不同的表型，提示呼吸事件和上气道结构可能与呻吟的病因有关，并对呻吟的治疗有一定意义。

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来源期刊

Nature and Science of Sleep Neuroscience-Behavioral Neuroscience

CiteScore

5.70

自引率

5.90%

发文量

245

审稿时长

16 weeks

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.

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