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Redalyc.CPAP Treatment for Catathrenia Revista Portuguesa de Pneumología ISSN: 0873-2159 [email protected] Sociedade Portuguesa de Pneumologia Portugal Dias, C.; Sousa, L.; Batata, L.; Teixeira, F.; Moita, J.; Moutinho dos Santos, J. CPAP treatment for catathrenia Revista Portuguesa de Pneumología, vol. 23, núm. 2, marzo-abril, 2017, pp. 101-104 Sociedade Portuguesa de Pneumologia Lisboa, Portugal Available in: http://www.redalyc.org/articulo.oa?id=169750147009 How to cite Complete issue Scientific Information System More information about this article Network of Scientific Journals from Latin America, the Caribbean, Spain and Portugal Journal's homepage in redalyc.org Non-profit academic project, developed under the open access initiative Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato. RESEARCH LETTERS 101 Conflicts of interest randomised controlled trial of efficacy, feasibility and costs. Respir Med. 2014;108:1387---95. The authors have no conflicts of interest to declare. J.C. Winck a,∗, J. Chaves Caminha b a References Faculdade de Medicina da Universidade do Porto, Portugal b Instituto de Ciências Biomédicas Abel Salazar, 1. Nava S. Behind a mask: tricks, pitfalls and preju- Universidade do Porto, Portugal dices for non invasive ventilation. Respir Care. 2013;58: 1367---76. ∗ Corresponding author. 2. Köhnlein T, Windisch W, Köhler D, Drabik A, Geiseler J, Hartl S, E-mail address: [email protected] (J.C. Winck). et al. Non-invasive positive pressure ventilation for the treat- ment of severe stable chronic obstructive pulmonary disease: a http://dx.doi.org/10.1016/j.rppnen.2015.12.008 prospective, multicentre, randomised, controlled clinical trial. 2173-5115/ Lancet Respir Med. 2014;2:698---705. © 2016 Sociedade Portuguesa de Pneumologia. Published by Elsevier 3. Hazenberg A, Kerstjens HA, Prins SC, Vermeulen KM, Wijk- Espa na,˜ S.L.U. This is an open access article under the CC BY-NC-ND stra PJ. Initiation of home mechanical ventilation at home: a license (http://creativecommons.org/licenses/by-nc-nd/4.0/ ). CPAP treatment for catathrenia home with the established pressure. The patients were eval- uated at 1 and 3 months of CPAP therapy, in the presence of Catathrenia is a rare, idiopathic sleep disorder classified as their bed partner. During the evaluation, patients were ques- an isolated symptom of sleep-disordered breathing (SDB). 1 tioned how they would classify their daytime complaints Its prevalence is unknown, 2 and its onset is usually in improvement (scale 0---100). After the first 3 months of adolescence. 3 Affected individuals are frequently unaware CPAP therapy, they were contacted monthly by telephone of their problem, and family members or bed partners to evaluate improvements over the remaining 3 months of commonly report strange sounds while breathing during therapy. sleep. 1,3 Eight patients were included. Five patients had abnor- The hallmark of this disorder is a deep inspiration mal sleepiness (ESS > 10). Six patients had symptoms of followed by prolonged expiration and a monotonous vocal- anxiety disorder and 7 were medicated with psycopharms ization resembling groaning, usually during rapid eye (Table 1). movement sleep. 1 Catathrenia events during the diagnostic and CPAP titra- No pharmacological treatments are available, 4 but some tion polysomnograms are displayed on Table 2. Diagnostic studies have shown partial or complete resolution of events PSG showed a mean respiratory disturbance index (RDI) with continuous positive airway pressure (CPAP) therapy, of 2.8 ± 3.29 events/h, a mean apnea---hypopnea index of especially in patients with a SDB associated. 2,4,5 1.2 ± 1.5 events/h. The mean number of catathrenia events The purpose of this study was to assess the effectiveness was 39.3 ± 26.6. Patient #6 experienced a partial resolution of CPAP treatment and a 6-month CPAP therapy in patients of events, despite an increase in pressure up to 12 cmH 2O. with catathrenia without SDB events associated. This patient did not tolerate a higher pressure. We performed a prospective study of patients with After 1 month of therapy, seven patients had signifi- catathrenia, diagnosed between 2008 and 2014, who under- cantly fewer moaning/groaning episodes and patient #6 went a CPAP titration PSG and subsequently initiated home reported a moderate reduction. After 3 months, seven CPAP therapy for 6 months. There were no exclusion patients reported complete resolution of nocturnal episodes criteria. and patient #6 reported maintenance of the moderate During anamnesis, the evaluated symptoms were: groan- reduction achieved with the first month of therapy. An ing, snoring, choking, apnea, daytime sleepiness (Epworth improvement in daytime complaints was reported by all Sleepiness Scale [ESS]), headache, fatigue, and anxi- patients (mean subjective improvement of 80/100). One ety/depression. The diagnosis was based on an overnight patient maintained an abnormal sleepiness (ESS > 10). Com- polysomnogram (PSG). A catathrenia event was defined as plete resolution of events was maintained during the 6 a deep inhalation followed by prolonged exhalation, and months of follow-up, except for patient #6 who continued to a monotonous vocalization resembling groaning. 1 After the exhibit partial improvement. This patient was subsequently diagnostic study, patients underwent a CPAP titration PSG treated with clonazepam for 6 months, but showed no addi- to correct catathrenia events. CPAP began at 4 cmH 2O and tional improvements. was progressively increased according to the type of respira- Although CPAP titration has been used in previous studies tory events observed. In the presence of obstructive events, of catathrenia, its goal was to correct apnea, hypopnea, and such as obstructive apnea or hypopnea, the pressure was flow limitation. 2,5 Other studies have reported an improve- increased by 2 cmH 2O every 15 min, and in the presence of ment in moaning/groaning with CPAP, but the majority of snoring, respiratory effort-related arousals, and/or groan- patients had associated SDB. 2,4 6 ing episodes, it was increased by 1 cmH 2O every 15 min, Iriarte et al. have argued that the pathological mech- until the events reduced in number or disappeared. Once anism underlying catathrenia is mainly obstructive. Our the optimal pressure was achieved, CPAP was initiated at results support this theory, as our patients responded to Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato. 102 Table 1 Demographic and clinical characteristics of patients. Patient Sex Age (years) BMI (kg/m 2) Chief Onset of Other Craniofacial Psycopharms ESS score complaint groaning symptoms examination 1 F 40 21.4 Groaning, Childhood Choking, Mallampati I, Paroxetine 13 excessive morning high narrow daytime headache, hard palate sleepiness panic attack, 2 F 25 27.7 Groaning, Childhood Morning Mallampati I, Ethyl 14 fatigue headache, deviated nasal loflazepate, panic attack septum trazodone 3 F 37 39.5 Groaning, 15 years of Depression Mallampati II, Paroxetine, 20 snoring, age long uvula Lorazepam, choking Trazodone 4 F 30 22 Groaning Childhood Tension Mallampati II, Cloxazolam 11 headache tonsillectomy 5 M 31 26 Groaning 25 years of Morning Mallampati II, --- 4 age headache and long uvula fatigue 6 M 32 27.2 Groaning, 28 years of Apnea, Mallampati I, Sertraline, 4 morning age depression High narrow alprazolan fatigue and hard palate headache 7 F 36 45.8 Groaning 26 years of Headache, Mallampati I Alprazolam 3 age fatigue, depression 8 F 34 18.2 Groaning 32 years of Choking, Mallampati I, Fluoxetine, 14 age morning high narrow alprazolam fatigue, hard palate, anxiety short uvula F --- female; M --- male; BMI --- body mass index; ESS --- Epworth Sleepiness Scale. RESEARCH LETTERS Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato. RESEARCH LETTERS 103 positive airway pressure. Vetrugno et al. 3 described a series of 10 patients with catathrenia and a normal RDI (as CPAP our patients) who had a post-inspiratory positive rise in 7 7 5 4 5 2 ESS After 12 10 endoesophageal pressure during events, higher than that movement; observed in expiration in eupnoic breathing, suggesting eye an expiratory upper airway obstruction. One possible CPAP mechanism that might explain the response observed in our 3 rapid patients is subtotal closure of the glottis during expiration. 0 0 0 0 0 0 0 (s) Maximum duration under 14 --- Our patients presented some differences in relation to REM previous published series: there was a preponderance of CPAP women; OSA was absent and the majority of patients were time; sleepy. It is possible that patients with more severe SDB, 0 0 0 0 0 8 0 0 (s) Minimum duration under PSG catathrenia events were unnoticed or obscured by other sleep respiratory sounds and therefore not detected. This may CPAP explain the lower prevalence in men and patients with Titration total OSA in our series. The higher frequency of young women --- 0 0 0 0 0 9 0 0 Catathrenia events under (total) is also probably related to the less severe SDB. As for TST sleepiness, it may be argued that it may be related to the O) concomitant psychiatric disorders and medications. Other CPAP 2 during index; sleepiness causes, recognized to contribute to sleep depri- used Peak 7 6 6 8 9 8 7 titration (cmH sound. 12 vation, were not analyzed in the present study. However, its improvement following efficacious catathrenia treat- 0.2 0.4 6.1 3.1 7 0.3 5 4.8 RDI ment suggest an association between both complaints and groaning s) warrants further research. While no clear association has a been demonstrated between catathrenia and psychiatric apnea/hypopnea 1 0 disorders, they marked a bold presence in our study (7/8 --- Clusters duration (mean, 59 23 15 76 56 80 67 patients).
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