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Published on: March 27, 2012
Obstructive events in children with Prader-Willi syndrome occur predominantly during rapid eye movement sleep
Anastasia Polytarchou1, Georgia Katsouli1, Marina Tsaoussoglou1
1Division of Pediatric Pulmonology and Sleep Disorders Laboratory, First Department of Pediatrics, National and Kapodistrian University of Athens School of Medicine and Aghia Sophia Children's Hospital, Athens, Greece.
Insights
Children with Prader-Willi syndrome (PWS) experience significantly more obstructive sleep apnea events during REM sleep than non-PWS children. This highlights the need for tailored sleep apnea assessments in PWS patients.
Area of Science:
- Pediatric Pulmonology
- Sleep Medicine
- Genetics
Background:
- Prader-Willi syndrome (PWS) is associated with a high incidence of obstructive sleep apnea syndrome (OSAS).
- Typically developing children with OSAS exhibit more obstructive events during REM sleep than NREM sleep.
- It is hypothesized that PWS patients have a greater propensity for REM sleep-related obstructive events.
Purpose of the Study:
- To compare the severity of obstructive sleep apnea (OSAS) during REM sleep between children with PWS and typically developing children.
- To investigate if PWS diagnosis is independently associated with increased REM sleep obstructive events.
Main Methods:
- Polysomnographic data from 12 children with PWS and 53 controls with OSAS were analyzed.
- Obstructive apnea-hypopnea index (OAHI) during NREM (OAHInrem) and REM (OAHIrem) sleep, and the OAHIrem/OAHI ratio were compared.
- A general linear model was used to adjust for confounders like age, gender, and obesity.
Main Results:
- Children with PWS and controls had similar overall OAHI and OAHInrem.
- PWS patients exhibited significantly higher OAHIrem (17.6 vs. 5 episodes/h) and OAHIrem/OAHI ratio (2.3 vs. 1) compared to controls.
- The association between PWS and increased OAHIrem/OAHI remained significant after adjusting for confounders.
Conclusions:
- In children with PWS, total sleep time OAHI underestimates OSAS severity during REM sleep.
- OSAS severity during REM sleep in PWS patients can be substantially higher, potentially twice that of controls.
- Mild OSAS detected without sleep staging in PWS may indicate moderate-to-severe REM sleep-related events.
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.
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