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Sleep-Related Catathrenia: An Enigma
October 2012, Vol 142, No. 4_MeetingAbstracts Signs and Symptoms of Chest Diseases | October 2012
Sleep-Related Catathrenia: An Enigma Yong Han Kim*, MD; Paramjeet Atwal, MD; Michael Weinstein, MD; Mary Cataletto, MD Winthrop University Hospital - Winthrop Sleep Disorders Center, Garden City, NY
Chest. 2012;142(4_MeetingAbstracts):1028A. doi:10.1378/chest.1389913
Abstract SESSION TYPE: Miscellaneous Cases II PRESENTED ON: Tuesday, October 23, 2012 at 11:15 AM - 12:30 PM INTRODUCTION: Catathrenia, derived from the Greek word (kata-like, threnia-to lament), is a sleep-related respiratory condition described as high pitched, monotonous groaning, moaning, respiratory dysrhythmia with bradypnea and vocalisation or inarticulate expiratory phonation. Clearly a rare disorder, its actual incidence and prevalence are unknown with a limited number of cases presented in the literature. CASE PRESENTATION: A fourteen year old boy with a history of bronchial asthma presented for evaluation of snoring and witnessed apneic episodes. The mother described the snoring as “funny noises” during sleep after apparent breath holding which awakens him at night. There was no prior history of sleeprelated breathing disorder or parasomnia. Physical exam revealed normal vital signs with BMI at 85th percentile for age. Oropharyngeal exam revealed a Mallampati class I airway and tonsils grade 2+. Overnight polysomnogram (PSG) revealed monotonous groaning sounds (Video / Image 1) heard during non-rapid eye movement (NREM) sleep and rapid eye movement (REM) sleep which
occurred in clusters and ended with a grunt or an arousal. Oxyhemoglobin saturation levels at or above 95% were noted throughout the study. Although the PSG findings closely resembled central apneas, careful inspection of the PSG waveforms along with audio and video recordings revealed the protracted expiration with monotonous groaning preceded by deep inspiration, which is diagnostic of catathrenia. Patient is currently using nasal CPAP resulting in improvement in groaning episodes. DISCUSSION: The occurrence during sleep of bradypnea with prolonged expiration and sound generation continues to be an enigma. Although multiple mechanisms have been proposed, the precise pathophysiology of catathrenia remains elusive, the response to treatment ranging from medications to continuous positive airway pressure (CPAP) remains variable, and the long term prognosis remains unexplained. Central sleep apnea may be confused with these bradypneic episodes but preceding deep inspiration and subsequent vocalizations in addition to the absence of oxygen desaturations differentiate these events from true central apneas. CONCLUSIONS: It is imperative not to confuse catathrenia with other respiratory and nonrespiratory disturbances during sleep for the important and obvious consequences on treatment and prognosis. 1) Vetrugno R, Lugares I, Ferini-Strambi L, Montagna P. Catathrenia (nocturnal groaning); What is it? Sleep 2008 Mar; 31 (3): 308-9. 2) Guilleminault C, Hagen CC, Khaja AM. Catathrenia: parasomnia or uncommon feature of sleep disordered breathing? Sleep 2008 Jan; 31 (1): 132-9. 3) Siddiqui F, Walters AS, Chokroverty S. Sleep Med. 2008 May; 9 (4): 460-1. DISCLOSURE: The following authors have nothing to disclose: Yong Han Kim, Paramjeet Atwal, Michael Weinstein, Mary Cataletto No Product/Research Disclosure Information
Winthrop University Hospital - Winthrop Sleep Disorders Center, Garden City, NY
Sleep-Related Catathrenia: An Enigma Yong Han Kim*, MD; Paramjeet Atwal, MD; Michael Weinstein, MD; Mary Cataletto, MD Winthrop University Hospital - Winthrop Sleep Disorders Center, Garden City, NY
Chest. 2012;142(4_MeetingAbstracts):1028A. doi:10.1378/chest.1389913
Abstract SESSION TYPE: Miscellaneous Cases II PRESENTED ON: Tuesday, October 23, 2012 at 11:15 AM - 12:30 PM INTRODUCTION: Catathrenia, derived from the Greek word (kata-like, threnia-to lament), is a sleep-related respiratory condition described as high pitched, monotonous groaning, moaning, respiratory dysrhythmia with bradypnea and vocalisation or inarticulate expiratory phonation. Clearly a rare disorder, its actual incidence and prevalence are unknown with a limited number of cases presented in the literature. CASE PRESENTATION: A fourteen year old boy with a history of bronchial asthma presented for evaluation of snoring and witnessed apneic episodes. The mother described the snoring as “funny noises” during sleep after apparent breath holding which awakens him at night. There was no prior history of sleeprelated breathing disorder or parasomnia. Physical exam revealed normal vital signs with BMI at 85th percentile for age. Oropharyngeal exam revealed a Mallampati class I airway and tonsils grade 2+. Overnight polysomnogram (PSG) revealed monotonous groaning sounds (Video / Image 1) heard during non-rapid eye movement (NREM) sleep and rapid eye movement (REM) sleep which
occurred in clusters and ended with a grunt or an arousal. Oxyhemoglobin saturation levels at or above 95% were noted throughout the study. Although the PSG findings closely resembled central apneas, careful inspection of the PSG waveforms along with audio and video recordings revealed the protracted expiration with monotonous groaning preceded by deep inspiration, which is diagnostic of catathrenia. Patient is currently using nasal CPAP resulting in improvement in groaning episodes. DISCUSSION: The occurrence during sleep of bradypnea with prolonged expiration and sound generation continues to be an enigma. Although multiple mechanisms have been proposed, the precise pathophysiology of catathrenia remains elusive, the response to treatment ranging from medications to continuous positive airway pressure (CPAP) remains variable, and the long term prognosis remains unexplained. Central sleep apnea may be confused with these bradypneic episodes but preceding deep inspiration and subsequent vocalizations in addition to the absence of oxygen desaturations differentiate these events from true central apneas. CONCLUSIONS: It is imperative not to confuse catathrenia with other respiratory and nonrespiratory disturbances during sleep for the important and obvious consequences on treatment and prognosis. 1) Vetrugno R, Lugares I, Ferini-Strambi L, Montagna P. Catathrenia (nocturnal groaning); What is it? Sleep 2008 Mar; 31 (3): 308-9. 2) Guilleminault C, Hagen CC, Khaja AM. Catathrenia: parasomnia or uncommon feature of sleep disordered breathing? Sleep 2008 Jan; 31 (1): 132-9. 3) Siddiqui F, Walters AS, Chokroverty S. Sleep Med. 2008 May; 9 (4): 460-1. DISCLOSURE: The following authors have nothing to disclose: Yong Han Kim, Paramjeet Atwal, Michael Weinstein, Mary Cataletto No Product/Research Disclosure Information
Winthrop University Hospital - Winthrop Sleep Disorders Center, Garden City, NY