The Value of a Well-Trained Ear: Incidental Detection of Catathrenia on Home Sleep Apnea Tests in Patients With Low Probability for Obstructive Sleep Apnea

Abstract

Catathrenia is a rare sleep-related breathing disorder marked by nocturnal groaning during prolonged exhalation. Home sleep apnea tests are growing in popularity, but no prior case reports exist documenting catathrenia detected by a home sleep apnea test. We report two cases of catathrenia incidentally detected in patients with low to intermediate risk of obstructive sleep apnea, in which diagnostic polysomnography was recommended but denied by insurers. Events were automatically scored as central apneas, leading to diagnostic uncertainty. The key to identification of catathrenia in both patients was combination of audio review of events, pattern of expiratory air trapping, and mismatch between central apnea flow signal attenuation and increased sound noted on audio amplitude signal. Diagnostic polysomnography was ordered in both cases for confirmation of diagnosis, leading to increased care costs. Keywords: catathrenia, central apnea, home sleep apnea test, parasomnia, sleep-related breathing disorder Citation: Kazaglis L. The value of a well-trained ear: incidental detection of catathrenia on home sleep apnea tests in patients with low probability for obstructive sleep apnea.

The clinical characteristic of catathrenia: a new look at an old issue—a systematic review of existing literature

“…In literature, catathrenia has been described as a condition in serious diseases such as fatal insomnia [44], Pitt-Hopkins syndrome [52], or mental retardation [37]. In our analysis, we determined that the majority of the comorbidities was made up of general disorders, such as allergic rhinitis, depression, attention-deficit hyperactivity disorder (ADHD), or obesity [16,37,45]. Our review also determined that comorbidities (excluding sleep disorders) played a role in the presentation of groaning during the NREM sleep; they shortened the minimal and maximal episode duration and scored less points in ESS.…”

“…Thirty-one articles [16-19, 22, 25-28, 30-33, 37-54] were included for further analysis. These included 6 cohort studies [16,26,31,[42][43][44], 1 case-control study [39], 10 case series [19,22,24,25,37,38,40,41,45,47], and 14 case reports [17,18,27,28,30,32,33,46,[49][50][51][52][53][54]. However, in 3 studies [16,25,43], data was reported with SD and mean values.…”

“…The remaining case reports [18,33,53,54] were determined to have a moderate risk of bias. Similarly, the majority of the case series publications (6 out of 10) was deemed to have a high risk of bias [22,24,37,40,45,47]. The remaining 4 papers had an overall moderate risk of bias [19,25,38,41].…”

“…Detailed descriptions for the risk of bias assessment have been presented in Table 4 for case reports, Table 5 for case series, and Table 6 for observational (cohort and case-control) studies. [33] No Yes Yes Yes Yes Yes No Yes Moderate Ramar K et [30] No Yes No Yes Yes No No Yes High Argollo NS et al [18] No Yes Yes Yes Yes Yes No Yes Moderate Manconi M et al [49] Yes Yes Yes Yes Yes Yes No Yes Low Grigg-Damberger M et al [50] No Yes Yes Yes No No No Yes High Romigi A et al [32] No Yes Yes Yes No No No No High Bansal R et al [51] Yes Yes Yes Yes No No No Yes High Motojima T et al [52] No Yes No Yes No No No Yes High Gómez T et al [53] No Yes Yes Yes Yes Yes No Yes Moderate Siddiqui F et al [17] No Yes Yes Yes Yes No No No High Bar C et al [28] No Yes Yes Yes Yes No No Yes High Carbajal-Mamani S et al [54] No Yes Yes Yes Yes Yes No Yes Moderate [41] No Yes Yes Yes Yes Yes Yes No No Yes Moderate Vetrugno R et al [22] No No Yes No No No Yes No No No High Kazaglis L et al [45] No Yes Yes No No Yes Yes No No No High Iriarte J et al [48] No Yes Yes No No No Yes No No Yes High…”

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

“…Previous studies have pointed out that groaning events, with a flat airflow signal similar to apneas, could be misclassified in home sleep apnea tests (HSAT) and PSG. 58 , 59 Therefore, catathrenia might be common in patients with OSA and has been underdiagnosed. These assumptions considering associations between catathrenia and OSA, need future investigations.…”