Obstructive events in children with Prader-Willi syndrome occur predominantly during rapid eye movement sleep

Abstract

Objective: Children with Prader-Willi syndrome (PWS) have a high prevalence of obstructive sleep apnea syndrome (OSAS). In most typically developing children with OSAS, more obstructive apneas and hypopneas occur during rapid eye movement (REM) than during non-REM (NREM) sleep. It was hypothesized that patients with PWS are even more prone to obstructive events in REM sleep than otherwise healthy subjects with OSAS.

Methods: Polysomnographic data of patients with PWS and of typically developing children (controls) with OSAS (apnea-hypopnea index [AHI] > 1 episode/h) were analyzed. The two groups were compared regarding obstructive AHI (OAHI), OAHI during NREM sleep (OAHInrem), OAHI during REM sleep (OAHIrem), and the OAHIrem/OAHI ratio (outcome measures). The association between PWS diagnosis and OAHIrem/OAHI was adjusted for confounders using a general linear model.

Results: Twelve children with PWS (median age 7.1 years [interquartile range 3.5, 12.4 years]) and 53 controls (6.5 years [3.9, 8.7 years]) were studied. Children with PWS and controls were similar regarding OAHI (p = 0.21) and OAHInrem (p = 0.76). However, subjects with PWS had higher OAHIrem (17.6 episodes/h [5.8, 25.8 episodes/h]) and OAHIrem/OAHI (2.3 [1.5, 3.2]) than controls (5 episodes/h [1.5, 8.1 episodes/h]; p = 0.002 and 1 [0.5, 2]; p = 0.003, respectively). The association between PWS diagnosis and higher OAHIrem/OAHI persisted after adjustment for age, gender, and obesity (p = 0.009).

Conclusion: In children with PWS, OAHI calculated for total sleep time does not reflect OSAS severity during REM sleep, which on average can be twice as high. Mild OSAS in patients with PWS demonstrated by polygraphy without sleep staging may correspond to a moderately-to-severely increased OAHIrem.

Keywords: Obstructive sleep apnea syndrome; Prader–Willi syndrome; REM sleep; Sleep hypopnea.