Πέμπτη 21 Ιουλίου 2016

Catathrenia, a REM predominant disorder of arousal?

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Publication date: Available online 20 July 2016
Source:Sleep Medicine
Author(s): Panagis Drakatos, Sean Higgins, Iain Duncan, Sara Stevens, Sakina Dastagir, Adam Birdseye, Dimosthenis Lykouras, Rexford Muza, Nadia Gildeh, Ivana Rosenzweig, Adrian J Williams, Guy D Leschziner, Brian D Kent
ObjectivesCatathrenia is an uncommon and poorly understood disorder, characterized by groaning during sleep occurring in tandem with prolonged expiration. Its classification, pathogenesis and clinical relevance remain debated, substantially due to the limited number of cases reported to date. We report a series of consecutive cases of catathrenia, their clinical and polysomnographic characteristics, and their subsequent management.MethodsConsecutive patients with catathrenia who had undergone full polysomnography in our institution over a 5-and-half-year period were included. Catathrenia events (CEs) were examined in clusters, formulating catathrenia periods (CPs). The relationships between CPs, sleep stage distribution, EEG arousals, and other sleep parameters were assessed, along with the clinical presentation and management of catathrenic patients.ResultsA total of 427 CPs were identified in 38 patients, 81% arising from REM sleep. EEG arousals preceded or coincided with the onset of 84% of CPs, which were of longer duration than those not associated with an arousal (57.3±56.8 vs 32.2±29.4 sec, p<0.001). Each CE had a characteristic airflow signal, with inspiration preceding a protracted expiration & a brief more rapid exhalation, followed by deep inspiration. Although the majority of patients were referred on the basis of bed-partner complaints, 44.7% complained of daytime sleepiness. CPAP and sleep-consolidating pharmacotherapy lead to subjective improvement, but were limited by poor long-term adherence.ConclusionsIn the largest series of catathrenia patients reported to date, we found that this rare disorder is characterized by a distinct breathing pattern, and arises predominantly from REM sleep, with arousals almost uniformly preceding or coinciding with the onset of catathrenia periods.



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