The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome

Background: Aortic dissection is a life-threatening manifestation of Marfan's syndrome. Preliminary evidence suggests that obstructive sleep apnea (OSA) is associated with aortic disease in Marfan's syndrome. Objectives: To study the effect of OSA on aortic events in Marfan's syndrome...

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Main Authors: Kohler, M, Senn, O, Pitcher, A, Forfar, C, Blair, E, Risby, P, Stradling, JR, Wordsworth, B
Format: Journal article
Published: 2013
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author Kohler, M
Senn, O
Pitcher, A
Forfar, C
Blair, E
Risby, P
Stradling, JR
Wordsworth, B
author_facet Kohler, M
Senn, O
Pitcher, A
Forfar, C
Blair, E
Risby, P
Stradling, JR
Wordsworth, B
author_sort Kohler, M
collection OXFORD
description Background: Aortic dissection is a life-threatening manifestation of Marfan's syndrome. Preliminary evidence suggests that obstructive sleep apnea (OSA) is associated with aortic disease in Marfan's syndrome. Objectives: To study the effect of OSA on aortic events in Marfan's syndrome. Methods: In patients with Marfan's syndrome, a sleep study was performed at baseline and OSA was defined as >5 events of apnea/hypopnea (A+H) per hour in bed. Operation because of progressive aortic dilatation and death because of aortic rupture were defined as 'aortic events'. Kaplan-Meier survival analyses were used to compare event-free survival in patients with and without OSA. Cox regression models were used to explore the effects of covariates on event-free survival. Results: Data from 44 patients (mean age 37.4 years, 30 females) were available for analysis; 15 patients (34.1%) had OSA. The median follow-up time was 29 (interquartile range 24-36) months. Five patients had an aortic event within the follow-up time. Median event-free survival was 51.6 months. Event-free survival was significantly shorter in patients with OSA compared to patients without OSA (p = 0.012). In univariate analysis, A+H was associated with aortic events [hazard ratio (HR) 1.09, 95% confidence interval (CI) 1.01-1.18, p = 0.023]. Taking the interaction between BMI and A+H into account increased the HR for A+H (HR 1.75, 95% CI 1.003-3.048, p = 0.049). This association was no longer significant when other covariates were forced into the multivariate analysis. Conclusions: These data suggest that aortic event-free survival may be shorter in patients with Marfan's syndrome and OSA compared to patients without OSA, but more data from well-designed studies are needed to prove this association. Copyright © 2012 S. Karger AG, Basel.
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spelling oxford-uuid:f8894bd7-0422-4bc0-89f3-95a1844077d72022-03-27T12:51:00ZThe impact of obstructive sleep apnea on aortic disease in Marfan's syndromeJournal articlehttp://purl.org/coar/resource_type/c_dcae04bcuuid:f8894bd7-0422-4bc0-89f3-95a1844077d7Symplectic Elements at Oxford2013Kohler, MSenn, OPitcher, AForfar, CBlair, ERisby, PStradling, JRWordsworth, BBackground: Aortic dissection is a life-threatening manifestation of Marfan's syndrome. Preliminary evidence suggests that obstructive sleep apnea (OSA) is associated with aortic disease in Marfan's syndrome. Objectives: To study the effect of OSA on aortic events in Marfan's syndrome. Methods: In patients with Marfan's syndrome, a sleep study was performed at baseline and OSA was defined as >5 events of apnea/hypopnea (A+H) per hour in bed. Operation because of progressive aortic dilatation and death because of aortic rupture were defined as 'aortic events'. Kaplan-Meier survival analyses were used to compare event-free survival in patients with and without OSA. Cox regression models were used to explore the effects of covariates on event-free survival. Results: Data from 44 patients (mean age 37.4 years, 30 females) were available for analysis; 15 patients (34.1%) had OSA. The median follow-up time was 29 (interquartile range 24-36) months. Five patients had an aortic event within the follow-up time. Median event-free survival was 51.6 months. Event-free survival was significantly shorter in patients with OSA compared to patients without OSA (p = 0.012). In univariate analysis, A+H was associated with aortic events [hazard ratio (HR) 1.09, 95% confidence interval (CI) 1.01-1.18, p = 0.023]. Taking the interaction between BMI and A+H into account increased the HR for A+H (HR 1.75, 95% CI 1.003-3.048, p = 0.049). This association was no longer significant when other covariates were forced into the multivariate analysis. Conclusions: These data suggest that aortic event-free survival may be shorter in patients with Marfan's syndrome and OSA compared to patients without OSA, but more data from well-designed studies are needed to prove this association. Copyright © 2012 S. Karger AG, Basel.
spellingShingle Kohler, M
Senn, O
Pitcher, A
Forfar, C
Blair, E
Risby, P
Stradling, JR
Wordsworth, B
The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title_full The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title_fullStr The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title_full_unstemmed The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title_short The impact of obstructive sleep apnea on aortic disease in Marfan's syndrome
title_sort impact of obstructive sleep apnea on aortic disease in marfan s syndrome
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