Oxygen Versus Medical Air for Treatment of CSA in Prader Will Syndrome

The Hospital for Sick Children

Status and phase: Completed; Phase 4

Conditions: Prader-Willi Syndrome; Sleep Apnea, Central

Treatments: Biological: Medical Air vs Oxygen

Study type: Interventional

Funder types: Other

Identifiers

NCT03031626

1000053577

Details and patient eligibility

About

The aim of this study is to determine if treatment with Medical Air (21% oxygen in room air) compared to supplemental oxygen (100% oxygen) will lead to similar improvements in the central apnea-hypopnea index (CAHI) for infants with Prader-Willi Syndrome.

Despite the vast amount of research investigating the cause of central sleep apnea, there remain gaps in knowledge, lending to further research efforts. The decision to compare oxygen to medical air is based on several theorized mechanisms. The first of which is the supposition that provision of medical air may act as an arousal stimulus for the hypothalamus, thereby preventing sleep disordered breathing. Secondly, the hypercapnic challenge performed by Livingston et al demonstrated a delayed hypercapneic arousal response in PWS subjects despite simultaneous hyperoxia, leading us to question if therapeutic oxygen really plays a significant role in treating CSA. Lastly, the delivery of medical air via nasal prongs may provide sufficient arousal to terminate the cycle of events leading to central apnea, as described by Urquhart et al.

A deeper understanding of central sleep apnea is essential to ameliorating its adverse sequelae, which include symptoms of ADHD, impaired attention, behavioral problems, and academic difficulties.

Enrollment: 9 patients

Sex: All

Ages: Under 2 years old

Volunteers: No Healthy Volunteers

Inclusion criteria

1. infants under age two with genetically confirmed Prader-Willi Syndrome
2. referred to HSC sleep clinic for evaluation with polysomnogram prior to initiation of growth hormone
3. infants found to have clinically significant central sleep apnea, defined as an apnea-hypopnea index (AHI) equal to or greater than 5

Exclusion criteria

1. infants delivered prematurely (less than 37 weeks gestational age)
2. term infants with a history of hypoxic-ischemic encephalopathy or stroke
3. any concurrent diagnoses that may cause sleep-disordered breathing (ie. craniofacial abnormalities, arnold-chiari malformation, etc)
4. infants with a need for daytime supplemental oxygen (ie. cardiac anomalies)
5. infants found to have low baseline oxygen saturations on PSG

Trial design

Primary purpose: Treatment

Allocation: Randomized

Interventional model: Crossover Assignment

Masking: Triple Blind

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9 participants in 2 patient groups

Arm A: Medical air followed by oxygen

Experimental group

Treatment:

Biological: Medical Air vs Oxygen

Arm B: Oxygen followed by medical air

Experimental group

Treatment:

Biological: Medical Air vs Oxygen