Prevalence and treatment of sleep-disordered breathing in Down's syndrome

ISRCTN	ISRCTN55685305
DOI	https://doi.org/10.1186/ISRCTN55685305
Protocol serial number	1
Sponsor	University of Edinburgh and NHS Lothian (UK) - Joint Sponsorship
Funders	Chief Scientist Office of the Scottish Executive Health Department (UK) (ref: CZH/4/549), Fondation Jerome Lejeune (France) (ref: R41361-195RSP), Added 08/10/2013:, Baily Thomas Charitable Trust (ref: TRUST/RNA/AC/TM/2634-5178)

Submission date: 20/12/2010
Registration date: 03/02/2011
Last edited: 27/10/2022

Recruitment status: No longer recruiting
Overall study status: Completed
Condition category: Respiratory

Prospectively registered

Protocol

Statistical analysis plan

Results

Individual participant data

Plain English summary of protocol

Not provided at time of registration

Contact information

Dr Renata Riha
Scientific

Department of Sleep Medicine
Royal Infirmary of Edinburgh
51 Little France Crescent
Edinburgh
EH16 4SA
United Kingdom

Phone	+44 (0)131 242 3882
Email	rlriha@hotmail.com

Study information

Primary study design	Interventional
Study design	Repeated measures parallel-arm controlled intention to treat study
Secondary study design	Randomised controlled trial
Study type	Participant information sheet
Scientific title	Controlled prospective trial of the effectiveness of continuous positive airway pressure therapy in adults with Down's syndrome
Study objectives	Continuous positive airway pressure (CPAP) use in Down's syndrome (DS) adults with obstructive sleep apnoea/hypopnoea syndrome (OSAHS)/sleep-disordered breathing (SDB) improves sleepiness and quality of life more effectively than lifestyle measures alone.
Ethics approval(s)	Scotland A Research Ethics Committee, 28/02/2011 and substantial amendment on 17/05/2011, ref: 11/MRE00/3
Health condition(s) or problem(s) studied	Sleep disordered breathing, obstructive sleep apnoea, Down's syndrome
Intervention	All participants with a diagnosis of sleep apnoea on home sleep study will be randomised to either the Lifestyle group or the CPAP Group. Participants allocated to the Lifestyle Group will receive written lifestyle advice on diet, exercise, sleep hygiene and sleeping position. Participants allocated to the CPAP Group will receive CPAP at the optimal pressure to treat their symptoms, along with written lifestyle advice as per the Lifestyle Group. Participants in both groups will be followed up at 1 week & at 1 month after randomisation. After 1 month, the Lifestyle Group will be offered CPAP, with additional follow ups at 1 week and 1 month after commencing CPAP. Both groups will then be followed at 3 months and 6 months after randomisation. At 6 months, participation in the study is complete, and all participants will be encouraged to remain on CPAP. Participants in both limbs will have anthropometric measures, cognitive function tests, health questionnaires, carer questionnaires and documentation of healthcare contact at baseline (randomisation), 1 month, 3 months & 6 months. All participants will complete and return a monthly diary recording health care contact, caffeine intake, medication, health questionnaires and a carer questionnaire.
Intervention type	Other
Primary outcome measure(s)	1. Change in Subjective Sleepiness: Epworth Sleepiness Scale (ESS) recorded at baseline, 1 week and monthly thereafter (averaged over 1, 3, and 6 months). 2. Change in health related quality of life: EQ-5D (measured at measured at baseline, 1, 3 and 6 months) valued using UK population tariffs. This will be used to estimate the cost per quality-adjusted life year (QALY) gained by providing CPAP in comparison to lifestyle measures. The analysis will incorporate health care utilisation, including hospital visits and GP visits during the trial (recorded by monthly diary) and 3 months prior to trial entry (obtained via GP letter and review of patient records).
Key secondary outcome measure(s)	1. Objective changes in emotional and behavioural function: Modified DBC-A - measured at baseline and monthly thereafter 2. Health status (quality of life): SF-12, SAQLI - baseline, 1, 3, 6 months 3. Cognitive function: All tests deleted, replaced with Arizona Cognitive Test Battery (ACTB) (Added 31/05/2011) 4. Adverse events: Side effects associated with CPAP usage, such as dry mouth - baseline and monthly thereafter, + 1 week after commencing treatment 5. Compliance with CPAP: Average hours of use per night from in-built machine timeclocks - baseline, 1, 3, 6 months 6. Carer burden: Modified CBI, GHQ-12, open-ended qualitative comments about the experience of caring - baseline and monthly thereafter Previous secondary outcome measures: 3. Cognitive function: Leiter-R, BPVSII, Raven Matrices, DSQIID - baseline, 1, 3, 6 months
Completion date	31/12/2014

Eligibility

Participant type(s)	Patient
Age group	Adult
Sex	All
Target sample size at registration	68
Total final enrolment	28
Key inclusion criteria	For the prevalence study: 1. Age greater than or equal to 16 years, either sex 2. A clinical diagnosis of Down's syndrome Additionally, for CPAP evaluation: 3. A clinical diagnosis of OSAHS (greater than or equal to 10 obstructive apnoeas hour on multichannel sleep study and symptoms of excessive daytime sleepiness or Epworth Sleepiness Scale [ESS] greater than or equal to 9) 4. Ability to give informed consent and comply with protocol (participant and/or relative/carer, as appropriate)
Key exclusion criteria	1. Previous exposure to CPAP therapy 2. Arterial oxygen saturation less than 90% on room air 3. Participants with forced expiratory volume in one second (FEV1) less than 60% 4. Participants with chronic heart failure or recent myocardial infarction (heart attack) 5. Participants with known moderate or severe dementia 6. Participants with severe behavioural problems that would preclude sleep studies or CPAP treatment 7. Inability to give informed consent AND relative/carer unable or unwilling to give informed consent 8. Inability to comply with the protocol
Date of first enrolment	01/02/2011
Date of final enrolment	31/12/2014

Locations

Countries of recruitment

United Kingdom
Scotland

Study participating centre

Department of Sleep Medicine

The Royal Infirmary of Edinburgh
51 Little France Crescent
Old Dalkeith Road
Edinburgh
EH16 4SA
United Kingdom

Results and Publications

Individual participant data (IPD) Intention to share	No
IPD sharing plan summary	Not provided at time of registration
IPD sharing plan	Not provided at time of registration

Study outputs

Output type	Details	Date created	Date added	Peer reviewed?	Patient-facing?
Results article		12/11/2020	27/10/2022	Yes	No
Other publications	Validation of pictorial Epworth sleepiness scale in adults with Down syndrome	01/02/2020	18/12/2020	Yes	No
Participant information sheet	Participant information sheet	11/11/2025	11/11/2025	No	Yes

Editorial Notes

27/10/2022: The following changes have been made:
1. Publication reference added.
2. The final enrolment number has been added from the reference.
18/12/2020: Publication reference added.
12/01/2017: No publications found in PubMed. Verifying study status with principal investigator.
08/10/2013: Anticipated end date was changed from 01/02/2013 to 31/12/2014.