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Catathrenia and Treatment With Positive Airway Pressure in the Pediatric Population

2019; American Academy of Sleep Medicine; Volume: 15; Issue: 12 Linguagem: Inglês

10.5664/jcsm.8100

1550-9397

Lacie Petitto, Gulnur Com, Rithea Jackson, Gresham T. Richter, Supriya Jambhekar,

Respiratory and Cough-Related Research

Free AccessCase ReportsCatathrenia and Treatment With Positive Airway Pressure in the Pediatric Population Lacie Petitto, APRN, DNP, Gulnur Com, MD, Rithea Jackson, RPSGT, Gresham Richter, MD, Supriya Jambhekar, MD Lacie Petitto, APRN, DNP *Address correspondence to: Lacie Petitto, Department of Pediatrics, Pulmonary/Sleep Division, Texas Children's Hospital, Houston, TX, 77094; Email: E-mail Address: [email protected] Arkansas Children's Sleep Disorders Center, Little Rock, Arkansas; , Gulnur Com, MD Department of Pediatrics, University of Arkansas for Medical Sciences, Little Rock, Arkansas , Rithea Jackson, RPSGT Arkansas Children's Sleep Disorders Center, Little Rock, Arkansas; , Gresham Richter, MD Department of Pediatrics, University of Arkansas for Medical Sciences, Little Rock, Arkansas , Supriya Jambhekar, MD Department of Pediatrics, University of Arkansas for Medical Sciences, Little Rock, Arkansas Published Online:December 15, 2019https://doi.org/10.5664/jcsm.8100Cited by:1SectionsAbstractPDF ShareShare onFacebookTwitterLinkedInRedditEmail ToolsAdd to favoritesDownload CitationsTrack Citations AboutABSTRACTCatathrenia, also known as sleep-related groaning, is a relatively rare sleep disorder with characteristics consistent with loud groaning on expiration during sleep. Organic causes of catathrenia are unknown and the decision of whether or not to treat is unclear. Limited research is available concerning appropriate treatment and none of the literature focuses primarily on pediatrics. We report a series of three, male pediatric cases with catathrenia that were treated with continuous positive airway pressure (CPAP) while identifying and comparing clinical symptoms, polysomnogram findings, treatment, and patient response to treatment. Catathrenia may be associated with abnormal nocturnal oxygenation and ventilation and may lead to negative clinical daytime symptoms which may warrant treatment. If catathrenia leads to sleep disruption and negative daytime symptoms, treatment with CPAP should be considered.Citation:Petitto L, Com G, Jackson R, Richter G, Jambhekar S. Catathrenia and treatment with positive airway pressure in the pediatric population. J Clin Sleep Med. 2019;15(12):1853–1857.INTRODUCTIONCatathrenia, also known as sleep-related groaning, is a rare disorder characterized by a chronic expiratory groan usually during rapid eye movement (REM) sleep according to the American Academy of Sleep Medicine (AASM).1 It is classified as a sleep-related breathing disorder and thought to be a combination of sleep-related breathing disorder and parasomnia.2Long-term effects of catathrenia are unknown and clinical significance is unclear.1 Due to the rarity of this disorder, there is limited literature concerning the diagnosis and treatment of catathrenia. A combination of oral surgeries (tonsillectomy, adenotonsillectomy, nasal turbinate reduction, septoplasty, pharyngoplasty) with or without oral appliances have been shown to improve symptoms in adults.3 Continuous positive airway pressure (CPAP) has been shown to be effective treatment and studied mainly in the adult population.3 The aims of this pediatric case series are: (1) Describe negative clinical effects; (2) identify coexisting adverse physiological findings (ie hypoxia, carbon dioxide retention, arousals); and (3) discuss need for treatment and effectiveness of treatment with use of CPAP therapy in a small group of children with catathrenia.REPORT OF CASESBetween May 2011 and October 2014, 3 pediatric patients presented to our multidisciplinary sleep center with complaints of moaning and groaning at night accompanied with daytime fatigue. All patients were male and their initial evaluations were conducted at the ages of 7, 12, and 14 years. Caregivers sought treatment for various reasons such as behavioral problems, disruption of sleep in those residing in the home, daytime somnolence, and morning headaches.All patients underwent an initial sleep clinic evaluation which included completion of the standard sleep clinic questionnaire. Medical charts were reviewed to obtain information regarding the patient's detailed sleep history, physical examination, and polysomnography (PSG) findings as this is a retrospective chart review. See Table 1 for details regarding patient description, PSG findings, treatment with CPAP, pressure settings, and outcomes.Table 1 Patient descriptions, clinical symptoms, treatment, and outcomes.Case 1Case 2Case 3Age, years7 (with mild developmental delay)1412Symptoms- Nocturnal groaning- Nocturnal moaning (worse in early morning)- Nocturnal groaning- Fatigue *- Fatigue *- Sleep disruptions- Fatigue *- Morning grogginess *- Behavioral problems *- Morning grogginess *- Parental anxiety because of groaning- No snoring- Family sleep disruption *- Morning headaches- No snoring- No snoringENT Examination- Normal examination- Normal examination- Normal examination- Tonsils absent- Tonsils not enlarged- Tonsils enlarged 2+Diagnostic PSG Summary †- Expiratory groaning with pauses- Expiratory groaning associated with arousal- Expiratory groaning with pauses- Sleep disruption- Mild oxygenation defect- Events equal between REM and NREM sleep- Bradycardia- Predominantly in REM sleep- Expiratory pauses- Hypoventilation- Predominantly in NREM sleepTitration PSGResolution of events and improved sleep architecture with CPAP pressure of 7 cmH2OResolution of events with pressure of 8 cmH2OResolution of events and normalized oxygenation with pressures of 4–6 cmH2O- mean ETCO2 46.1 mmHg- highest ETCO2 50.2 mmHg- 0 expiratory groaning events/hCPAP Pressure, fixed cmH2O786CPAP Adherence at 1 year- Total use: 92.3% of nights- Total use: 86% of nightsUnknown- Use for > 4 h/night: 89.7% of nights- Use for > 4 h/night: 77% of nights- Average nightly use: 8 hours, 2 minutes- Average nightly use: 6 hours, 43 minutesOutcome/Follow-upAll symptoms resolved with CPAP pressure of 10 cmH2OAll symptoms resolved with CPAP pressure of 8 cmH2OUnable to implement CPAP due to lack of insurance coverage of CPAP. Patient was lost to follow-up.* CPAP was initiated due to these findings which were deemed significant. † See Table 2 for details. CPAP = continuous positive airway pressure, ENT = ear, nose and throat, ETCO2 = end tidal carbon dioxide, NREM = non-rapid eye movement, PSG = polysomnography, REM = rapid eye movement, SpO2 = peripheral capillary oxygen saturation.Data were obtained utilizing the Sandman Elite 9.1 or 10.1.1 digital sleep system (Embla Systems, Broomfield, Colorado, United States). The following variables were recorded: Electroencephalogram (F3/A2, F4/A1, C3/A2, C4/A1, O1/A2, O2/A1) right and left electroculograms, submental electromyogram, tibial +/− upper extremity electromyograms, electrocardiogram, abdominal and chest respiratory effort (Sensormedics or Embla inductance plethysmography), end tidal CO2 (BCI capnograph), airflow (Dymedix or Embla thermistor), SpO2 (Nellcor or Nonin pulse oximetry), and nasal pressure monitoring (ProTech/Embla PTAF2). Continuous time-linked sound/visual recordings were obtained using audio and infrared technology. All studies were scored using the criteria in the 2007 or 2012 edition of The AASM Manual for the Scoring of Sleep and Associated Events: Rules, Terminology and Technical Specifications (as applicable).4,5A catathrenia event was scored when there was an expiratory groan sound associated with respiratory pause followed by either an arousal or a 3% oxygen desaturation. Due to its similarity in appearance to a central apnea on the PSG, it was scored as a central apnea and included in the apnea-hypopnea index in the initial PSG report. For the purpose of this manuscript, catathrenia events were reported separately. During CPAP titration, CPAP was initiated at 4 cm and titrated to resolve catathrenia events. See Figure 1, Figure 2, Figure 3 and Table 2 for PSG results.Figure 1: Case 1: 3-minute tracing.Sleep-related expiratory groaning (seen in snore channel) with respiratory pause during non-rapid eye movement sleep followed by arousal.Download FigureFigure 2: Case 2: 3-minute tracing.Sleep-related expiratory groaning (seen in snore channel) with respiratory pause during non-rapid eye movement sleep followed by arousal, associated with increased end-tidal carbon dioxide levels.Download FigureFigure 3: Case 3: 3-minute tracing.Sleep-related expiratory groaning (seen in chin electromyography channel, no snore channel available) with respiratory pause during rapid eye movement sleep followed by arousal, associated with oxygen desaturation.Download FigureTable 2 Diagnostic polysomnography results.ParameterCase 1Case 2Case 3Sleep efficiency (%)86.19772Stage N1 sleep (% TST)104.73.9Stage N2 sleep (% TST)45.448.953.5Stage N3 sleep (% TST)2625.628Stage R sleep (% TST)18.620.914.6Arousal index (events/h)22.413.73.6Central apnea index (events/h)1.21.10.7Obstructive apnea index (events/h)000CEI (events/h)12.85.81.4Hypopnea index (events/h)10.40.9AHI (events/h)2.21.51.6REI (AHI + CEI; events/h)14.17.33REM sleep-related REI (events/h)10.70.610.4REM sleep-related CEI (events/h)009.4PLM index (events/h)100Mean oxygen saturation (%)979694.7Oxygen saturation nadir (%)869388% TST with saturation less than 95%0.41737Mean ETCO2 (mmHg)37.249.545.6Maximum ETCO2 (mmHg)45.755.750.5% TST with ETCO2 > 50 mmHg033.60.1AHI = apnea-hypopnea index, CEI = catathrenia event index, ETCO2 = end tidal carbon dioxide, PLM = periodic limb movement, REI = respiratory event index, REM= rapid eye movement, TST = total sleep time.DISCUSSIONTo our knowledge, this is the first manuscript that focuses on catathrenia in children. The most recent social media survey and comprehensive literature review study of catathrenia described 47 patients, all of which were adults.6 There are few articles (including individual case reports) that include pediatric patients. The studies' primary focus was not the pediatric population and none described treatment of children with CPAP.7–13 Currently, there is no reported literature concerning diagnosis and treatment of catathrenia with a focus on children.In all three of our patients, catathrenia presented as moaning and groaning at night with various secondary daytime effects. All patients also had a complaint of daytime fatigue. This has also been reported in adult patients in other studies.3,9,10,14,15 In these adult patients, physiological consequences (oxygen desaturation, arousals, or hypoventilation) associated with catathrenia were found on all PSG reports. Nocturnal groaning/moaning, fatigue, poor sleep, family sleep disruption, morning grogginess, and behavioral problems were also concurrent clinical symptoms present in these cases. With CPAP treatment, nocturnal groaning, physiological abnormalities, and daytime symptoms resolved in two of our three patients. The third patient in this case series cannot be commented on as CPAP could not be initiated due to lack of insurance coverage for treatment. This patient was lost to follow up due to social reasons and hence other forms of treatment, ie surgery or oral appliances could not be attempted. These findings concur with studies in the adult population when CPAP was used for treatment.2,3,7,15,16 Other studies have reported limited to no success with resolution of catathrenia with CPAP.9,14CONCLUSIONSCPAP treatment can be considered for catathrenia if the PSG concludes negative physiologic consequences associated with catathrenia and/or if a patient is experiencing clinical symptoms affecting quality of life in children. CPAP treatment may resolve both sleep-related negative physiologic and clinical effects of catathrenia. Since no other clearcut effective treatments have been reported in the pediatric population, we suggest that CPAP be considered as an effective treatment option for catathrenia. Further research is necessary to expand knowledge of catathrenia, its negative consequences, and treatment including effectiveness of surgery with or without oral appliances and CPAP in the pediatric population.DISCLOSURE STATEMENTAll authors have seen and approved this manuscript for submission. Work for this study was performed at Arkansas Children's Hospital, Little Rock, AR. The authors report no conflicts of interest.ABBREVIATIONSAASMAmerican Academy of Sleep MedicineAHIapnea-hypopnea indexCPAPcontinuous positive airway pressurePSGpolysomnographyREMrapid eye movementSpO2peripheral capillary oxygen saturationREFERENCES1. American Academy of Sleep Medicine. International Classification of Sleep Disorders. 2014. 3rd ed.. Darien, IL: American Academy of Sleep Medicine. Google Scholar2. Ott SR, Hamacher J, Seifert E. Bringing light to the sirens of night: laryngoscopy in catathrenia during sleep. Eur Respir J. 2011;37(5):1288–1289. CrossrefGoogle Scholar3. 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CrossrefGoogle Scholar Previous article Next article FiguresReferencesRelatedDetailsCited byMandibular advancement device as treatment trial for catathrenia (nocturnal groaning)Yu M, Hao Z, Xu L, Wen Y, Han F and Gao X Journal of Clinical Sleep Medicine, Vol. 17, No. 9, (1767-1776), Online publication date: 1-Sep-2021. Volume 15 • Issue 12 • December 15, 2019ISSN (print): 1550-9389ISSN (online): 1550-9397Frequency: Monthly Metrics History Submitted for publicationApril 30, 2019Submitted in final revised formSeptember 3, 2019Accepted for publicationSeptember 4, 2019Published onlineDecember 15, 2019 Information© 2019 American Academy of Sleep MedicineKeywordscontinuous positive airway pressureCPAPpediatricsCatathreniaPDF download