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

Phase 4

Recruiting

• Conditions: Prader-Willi Syndrome; Sleep Apnea, Central
• Interventions: Biological: Medical Air vs Oxygen

• Registration Number: NCT03031626
• Lead Sponsor: The Hospital for Sick Children

Brief Summary

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.

Detailed Description

Not available

Study Timeline

• Study Start: 2016-09-01
• Study First Submitted: 2017-01-20
• Study First Submitted QC: 2017-01-23
• Study First Posted: 2017-01-25
• Status Verified: 2024-10
• Last Update Submitted: 2024-10-25
• Last Update Posted: 2024-10-28
• Primary Completion: 2024-12-15
• Study Completion: 2024-12-15

Recruitment & Eligibility

• Status: RECRUITING
• Sex: All
• Target Recruitment: 10

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

Study & Design

• Study Type: INTERVENTIONAL
• Study Design: CROSSOVER

Arm && Interventions

Group	Intervention	Description
Arm A: Medical air followed by oxygen	Medical Air vs Oxygen	-
Arm B: Oxygen followed by medical air	Medical Air vs Oxygen	-

Primary Outcome Measures

Name	Time	Method
Delta CAHI1	2 years	Difference in CAHI at baseline compared to supplemental oxygen Delta CAHI1: CAHIoxygen - CAHIbaseline
Difference in CAHI1 and CAHI2	2 years	A comparison of change in CAHI1 and change in CAHI2 DeltaCAHI1: DeltaCAHI2
Delta CAHI2	2 years	Difference in CAHI at baseline compared to medical air Delta CAHI2: CAHImedical air - CAHIbaseline

Secondary Outcome Measures

Name	Time	Method
Arousal Index1	2 years	Difference in Arousal Index at baseline compared to medical air Delta Arousal Index: Arousal Indexmedical air - Arousal Indexbaseline
Arousal Index2	2 years	Difference in Arousal Index at baseline compared to Supplemental oxygen Delta Arousal Index: Arousal Indexoxygen - Arousal Indexbaseline
Desaturation Index1	2 years	Difference in Desaturation Index at baseline compared to medical air Delta Arousal Index: Desaturation Indexmedical air - Desaturation Indexbaseline
Desaturation Index2	2 years	Difference in Desaturation Index at baseline compared to supplemental oxygen Delta Arousal Index: Desaturation Indexoxygen - Desaturation Indexbaseline

Trial Locations

Locations (1)

The Hospital for Sick Children; 🇨🇦 Toronto, Ontario, Canada