Obstructive sleep apnoea and quality of life in Ehlers-Danlos syndrome: a parallel cohort study.
- Gaisl T Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- Giunta C Division of Metabolism, University Children's Hospital Zurich, Zurich, Switzerland.
- Bratton DJ Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- Sutherland K Department of Respiratory and Sleep Medicine, Royal North Shore Hospital, Sydney, New South Wales, Australia.
- Schlatzer C Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- Sievi N Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- Franzen D Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- Cistulli PA Department of Respiratory and Sleep Medicine, Royal North Shore Hospital, Sydney, New South Wales, Australia.
- Rohrbach M Division of Metabolism, University Children's Hospital Zurich, Zurich, Switzerland.
- Kohler M Department of Pulmonology, University Hospital Zurich, Zurich, Switzerland.
- 2017-01-12
Published in:
- Thorax. - 2017
Clinical Epidemiology
Connective tissue disease associated lung disease
Sleep apnoea
Adult
Ehlers-Danlos Syndrome
Female
Follow-Up Studies
Humans
Male
Middle Aged
Polysomnography
Prevalence
Prospective Studies
Quality of Life
Risk Factors
Sleep Apnea, Obstructive
Switzerland
English
BACKGROUND
Patients with the connective tissue disorder Ehlers-Danlos syndrome (EDS) often suffer from fatigue, excessive daytime sleepiness and impaired quality of life. Obstructive sleep apnoea (OSA) may be an underlying cause for these symptoms but its prevalence in this population is unclear.
METHODS
In this prospective parallel-cohort study, we included 100 adult patients with EDS (46% hypermobile-type, 35% classical-type and 19% other), which were one-to-one matched to 100 healthy adult controls according to sex, age, weight and height. Participants underwent structured interviews (including short-form 36) and level-3 respiratory polygraphy. OSA was defined as apnoea-hypopnea index ≥5/hour. Photographic craniofacial phenotyping was conducted in a subgroup. Conditional logistic regression was used to compare the prevalence of OSA.
RESULTS
In patients with EDS, OSA prevalence was 32% versus 6% in the matched control group (OR 5.3 (95% CI 2.5 to 11.2); p<0.001). The EDS group reported impaired quality of life in all dimensions (p<0.05) and significantly higher excessive daytime sleepiness measured by the Epworth Sleepiness Scale (median (quartiles) 11 (7-14) vs 7 (5-10); p<0.001). OSA severity was positively associated with daytime sleepiness and lower quality of life in the EDS group. There was no evidence of a difference between the two study groups in terms of craniofacial phenotypes.
CONCLUSIONS
The prevalence of OSA is higher in patients with EDS than in a matched control group. This is of clinical relevance as it is associated with fatigue, excessive daytime sleepiness and impaired quality of life. Further studies are needed to assess the clinical benefit of OSA treatment in patients with EDS.
TRIAL REGISTRATION NUMBER
NCT02435745.
Patients with the connective tissue disorder Ehlers-Danlos syndrome (EDS) often suffer from fatigue, excessive daytime sleepiness and impaired quality of life. Obstructive sleep apnoea (OSA) may be an underlying cause for these symptoms but its prevalence in this population is unclear.
METHODS
In this prospective parallel-cohort study, we included 100 adult patients with EDS (46% hypermobile-type, 35% classical-type and 19% other), which were one-to-one matched to 100 healthy adult controls according to sex, age, weight and height. Participants underwent structured interviews (including short-form 36) and level-3 respiratory polygraphy. OSA was defined as apnoea-hypopnea index ≥5/hour. Photographic craniofacial phenotyping was conducted in a subgroup. Conditional logistic regression was used to compare the prevalence of OSA.
RESULTS
In patients with EDS, OSA prevalence was 32% versus 6% in the matched control group (OR 5.3 (95% CI 2.5 to 11.2); p<0.001). The EDS group reported impaired quality of life in all dimensions (p<0.05) and significantly higher excessive daytime sleepiness measured by the Epworth Sleepiness Scale (median (quartiles) 11 (7-14) vs 7 (5-10); p<0.001). OSA severity was positively associated with daytime sleepiness and lower quality of life in the EDS group. There was no evidence of a difference between the two study groups in terms of craniofacial phenotypes.
CONCLUSIONS
The prevalence of OSA is higher in patients with EDS than in a matched control group. This is of clinical relevance as it is associated with fatigue, excessive daytime sleepiness and impaired quality of life. Further studies are needed to assess the clinical benefit of OSA treatment in patients with EDS.
TRIAL REGISTRATION NUMBER
NCT02435745.
- Language
-
- English
- Open access status
- bronze
- Identifiers
-
- DOI 10.1136/thoraxjnl-2016-209560
- PMID 28073822
- Persistent URL
- https://sonar.ch/global/documents/47396
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