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Central sleep-disordered breathing and the effects of oxygen therapy in infants with Prader-Willi syndrome
D S Urquhart1, T Gulliver, G Williams
1Department of Paediatric Respiratory and Sleep Medicine, Mater Children's Hospital, South Brisbane, Queensland, Australia.
Insights
Infants with Prader-Willi Syndrome (PWS) often experience central sleep-disordered breathing. Supplemental oxygen (O2) therapy significantly improved breathing patterns and oxygen levels in these infants.
Area of Science:
- Pediatric Pulmonology
- Sleep Medicine
- Genetics
Background:
- Prader-Willi Syndrome (PWS) is associated with sleep-disordered breathing, including hypersomnolence and obstructive sleep apnea.
- Central sleep breathing abnormalities are present from infancy in children with PWS.
Purpose of the Study:
- To characterize breathing patterns in infants diagnosed with Prader-Willi Syndrome.
- To evaluate the impact of supplemental oxygen (O2) on these breathing patterns.
Main Methods:
- Retrospective cohort study involving infants with PWS.
- Full polysomnography was performed, with split-night studies in air and supplemental O2 for infants with frequent desaturations.
- Analysis of central apnea index (CAI), central event index (CEI), and oxygen saturation (SpO2).
Main Results:
- Ten infants with PWS underwent 30 split-night studies.
- Baseline median CAI was 4.7/hour, with associated SpO2 desaturations.
- Supplemental O2 therapy significantly reduced CAI to 2.5/hour (p=0.002) and improved SpO2, with no change in obstructive events.
Conclusions:
- Infants with PWS can exhibit central sleep-disordered breathing leading to frequent desaturations.
- Supplemental O2 therapy demonstrates efficacy in improving breathing parameters and oxygenation in this population.
- Further longitudinal studies are needed to determine the onset of obstructive symptoms in PWS patients.
Objectives:
To describe breathing patterns in infants with Prader-Willi Syndrome (PWS), as well as the effects of supplemental oxygen (O2) on breathing patterns. Children with PWS commonly have sleep-disordered breathing, including hypersomnolence and obstructive sleep apnoea, as well as central sleep breathing abnormalities that are present from infancy.
Design:
Retrospective cohort study.
Patients:
Infants with a diagnosis of PWS.
Setting:
Tertiary children's hospital.
Interventions:
Infants with PWS underwent full polysomnography, and in those with frequent desaturations associated with central events, supplemental O2 during sleep was started and followed with regular split-night studies (periods in both air and O2).
Results:
Thirty split-night studies on 10 infants (8 female) aged 0.06-1.79 (median 0.68, IQR 0.45, 1.07) years were undertaken. At baseline (ie, air), children with PWS had a median (IQR) central apnoea index (CAI) of 4.7 (1.9, 10.6) per hour, with accompanying falls in oxygen saturation (SpO2). O2 therapy led to statistically significant reductions in CAI to 2.5/hour (p=0.002), as well as a reduced central event index (CEI) and improved SpO2. No change in the number of obstructive events was noted. Central events were more prevalent in rapid-eye movement/active sleep.
Conclusions:
It is concluded that infants with PWS may have central sleep-disordered breathing, which, in some children, may cause frequent desaturations. Improvements in CAI and CEI as well as oxygenation were noted with O2 therapy. Longitudinal work with this patient group would help to establish the timing of onset of obstructive symptoms.
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