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Prevalence and Risk Factors of Sleep Disordered Breathing in Fabry disease: A Prospective Cohort Study
Excessive daytime sleepiness (EDS) is a frequently reported and not well-understood symptom in patients with Fabry disease (FD). Sleep-disordered breathing (SDB) is a possible factor. As deposition of glycosphingolipids in the upper airway muscles is likely, we hypothesized that obstructive sleep ap...
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| Published in: | Medicine (Baltimore) 2015-12, Vol.94 (52), p.e2413-e2413 |
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| creator | Franzen, Daniel Gerard, Nicolas Bratton, Daniel J. Wons, Annette Gaisl, Thomas Sievi, Noriane A. Clarenbach, Christian F. Kohler, Malcolm Krayenbühl, Pierre A. |
| description | Excessive daytime sleepiness (EDS) is a frequently reported and not well-understood symptom in patients with Fabry disease (FD). Sleep-disordered breathing (SDB) is a possible factor. As deposition of glycosphingolipids in the upper airway muscles is likely, we hypothesized that obstructive sleep apnoea (OSA) is highly prevalent in FD and positively associated with its severity.All patients with FD who are followed in the Fabry cohort of the University Hospital Zurich (n = 62) were asked to participate in this prospective cohort study. Eligible patients were prospectively investigated by assessing their daytime sleepiness using the Epworth Sleepiness Scale (ESS), the severity of FD using the Mainz Severity Score Index (MSSI), and by an ambulatory overnight respiratory polygraphy between November 1, 2013, and January 31, 2015. SDB was defined as an apnea/hypopnea index (AHI) of > 5/h.Fifty-two patients (mean ± SD age 42.8 ± 14.7 years, 33% men, mean ± SD BMI 23.4 ± 3.6 kg/m) with a median (IQR) MSSI of 12 (5-19) were included. Median (IQR) ESS was 6 (2-10) and 7 patients (14%) had an ESS > 10. Thirteen patients (25%) had SDB (78% obstructive sleep apnea, 22% central sleep apnea). In the multivariable analysis, the age was the only statistically significant predictor of SDB (OR 1.11, 95% CI 1.04-1.18, P = 0.001). ESS was associated with depression (P |
| doi_str_mv | 10.1097/MD.0000000000002413 |
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Sleep-disordered breathing (SDB) is a possible factor. As deposition of glycosphingolipids in the upper airway muscles is likely, we hypothesized that obstructive sleep apnoea (OSA) is highly prevalent in FD and positively associated with its severity.All patients with FD who are followed in the Fabry cohort of the University Hospital Zurich (n = 62) were asked to participate in this prospective cohort study. Eligible patients were prospectively investigated by assessing their daytime sleepiness using the Epworth Sleepiness Scale (ESS), the severity of FD using the Mainz Severity Score Index (MSSI), and by an ambulatory overnight respiratory polygraphy between November 1, 2013, and January 31, 2015. SDB was defined as an apnea/hypopnea index (AHI) of > 5/h.Fifty-two patients (mean ± SD age 42.8 ± 14.7 years, 33% men, mean ± SD BMI 23.4 ± 3.6 kg/m) with a median (IQR) MSSI of 12 (5-19) were included. Median (IQR) ESS was 6 (2-10) and 7 patients (14%) had an ESS > 10. Thirteen patients (25%) had SDB (78% obstructive sleep apnea, 22% central sleep apnea). In the multivariable analysis, the age was the only statistically significant predictor of SDB (OR 1.11, 95% CI 1.04-1.18, P = 0.001). ESS was associated with depression (P < 0.001) but not AHI nor age.This study shows that SDB, especially obstructive sleep apnea is highly prevalent in patients with Fabry disease. However, EDS in FD seems to be related with depression rather than SDB.ClinicalTrials.gov (identifier: NCT01947634).</description><identifier>ISSN: 0025-7974</identifier><identifier>EISSN: 1536-5964</identifier><identifier>DOI: 10.1097/MD.0000000000002413</identifier><identifier>PMID: 26717401</identifier><language>eng</language><publisher>United States: Wolters Kluwer Health, Inc. All rights reserved</publisher><subject>Adult ; Fabry Disease - complications ; Female ; Humans ; Male ; Observational Study ; Prevalence ; Prospective Studies ; Risk Factors ; Sleep Apnea Syndromes - epidemiology ; Sleep Apnea Syndromes - etiology ; Sleep Apnea, Central - epidemiology ; Sleep Apnea, Central - etiology ; Sleep Apnea, Obstructive - epidemiology ; Sleep Apnea, Obstructive - etiology</subject><ispartof>Medicine (Baltimore), 2015-12, Vol.94 (52), p.e2413-e2413</ispartof><rights>Wolters Kluwer Health, Inc. All rights reserved.</rights><rights>Copyright © 2015 Wolters Kluwer Health, Inc. All rights reserved. 2015</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c3550-2ab29fae0874b2fee1e2f94a8be97b08efce9607979a43c27f9c92c9afd63ded3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5291642/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5291642/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,727,780,784,885,27924,27925,53791,53793</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26717401$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Franzen, Daniel</creatorcontrib><creatorcontrib>Gerard, Nicolas</creatorcontrib><creatorcontrib>Bratton, Daniel J.</creatorcontrib><creatorcontrib>Wons, Annette</creatorcontrib><creatorcontrib>Gaisl, Thomas</creatorcontrib><creatorcontrib>Sievi, Noriane A.</creatorcontrib><creatorcontrib>Clarenbach, Christian F.</creatorcontrib><creatorcontrib>Kohler, Malcolm</creatorcontrib><creatorcontrib>Krayenbühl, Pierre A.</creatorcontrib><title>Prevalence and Risk Factors of Sleep Disordered Breathing in Fabry disease: A Prospective Cohort Study</title><title>Medicine (Baltimore)</title><addtitle>Medicine (Baltimore)</addtitle><description>Excessive daytime sleepiness (EDS) is a frequently reported and not well-understood symptom in patients with Fabry disease (FD). Sleep-disordered breathing (SDB) is a possible factor. As deposition of glycosphingolipids in the upper airway muscles is likely, we hypothesized that obstructive sleep apnoea (OSA) is highly prevalent in FD and positively associated with its severity.All patients with FD who are followed in the Fabry cohort of the University Hospital Zurich (n = 62) were asked to participate in this prospective cohort study. Eligible patients were prospectively investigated by assessing their daytime sleepiness using the Epworth Sleepiness Scale (ESS), the severity of FD using the Mainz Severity Score Index (MSSI), and by an ambulatory overnight respiratory polygraphy between November 1, 2013, and January 31, 2015. SDB was defined as an apnea/hypopnea index (AHI) of > 5/h.Fifty-two patients (mean ± SD age 42.8 ± 14.7 years, 33% men, mean ± SD BMI 23.4 ± 3.6 kg/m) with a median (IQR) MSSI of 12 (5-19) were included. Median (IQR) ESS was 6 (2-10) and 7 patients (14%) had an ESS > 10. Thirteen patients (25%) had SDB (78% obstructive sleep apnea, 22% central sleep apnea). In the multivariable analysis, the age was the only statistically significant predictor of SDB (OR 1.11, 95% CI 1.04-1.18, P = 0.001). ESS was associated with depression (P < 0.001) but not AHI nor age.This study shows that SDB, especially obstructive sleep apnea is highly prevalent in patients with Fabry disease. However, EDS in FD seems to be related with depression rather than SDB.ClinicalTrials.gov (identifier: NCT01947634).</description><subject>Adult</subject><subject>Fabry Disease - complications</subject><subject>Female</subject><subject>Humans</subject><subject>Male</subject><subject>Observational Study</subject><subject>Prevalence</subject><subject>Prospective Studies</subject><subject>Risk Factors</subject><subject>Sleep Apnea Syndromes - epidemiology</subject><subject>Sleep Apnea Syndromes - etiology</subject><subject>Sleep Apnea, Central - epidemiology</subject><subject>Sleep Apnea, Central - etiology</subject><subject>Sleep Apnea, Obstructive - epidemiology</subject><subject>Sleep Apnea, Obstructive - etiology</subject><issn>0025-7974</issn><issn>1536-5964</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><recordid>eNpdUU1v1DAQtRCILgu_AAn5yCXFX4ljDkhllwJSKyoKZ8uxx41pNt7azlb770nZUhXmMtLMmzfz5iH0mpJjSpR8d74-Jo-CCcqfoAWteVPVqhFP0WIu1pVUUhyhFzn_IoRyycRzdMQaSaUgdIH8RYKdGWC0gM3o8PeQr_GpsSWmjKPHlwPAFq9DjslBAoc_JjClD-MVDuMM7NIeu5DBZHiPT_BFinkLtoQd4FXsYyr4skxu_xI982bI8Oo-L9HP008_Vl-qs2-fv65OzirL65pUzHRMeQOklaJjHoAC80qYtgMlO9KCt6AaMktSRnDLpFdWMauMdw134PgSfTjwbqduA87CWJIZ9DaFjUl7HU3Q_3bG0OuruNM1U7QRbCZ4e0-Q4s0EuehNyBaGwYwQp6yprDkhLZ_fvET8ALWz6JzAP6yhRN85pM_X-n-H5qk3jy98mPlryQwQB8BtHAqkfD1Mt5B0D2Yo_R--WipWMUJrylhLqrsK4b8BxcSdQg</recordid><startdate>20151201</startdate><enddate>20151201</enddate><creator>Franzen, Daniel</creator><creator>Gerard, Nicolas</creator><creator>Bratton, Daniel J.</creator><creator>Wons, Annette</creator><creator>Gaisl, Thomas</creator><creator>Sievi, Noriane A.</creator><creator>Clarenbach, Christian F.</creator><creator>Kohler, Malcolm</creator><creator>Krayenbühl, Pierre A.</creator><general>Wolters Kluwer Health, Inc. All rights reserved</general><general>Wolters Kluwer Health</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20151201</creationdate><title>Prevalence and Risk Factors of Sleep Disordered Breathing in Fabry disease: A Prospective Cohort Study</title><author>Franzen, Daniel ; Gerard, Nicolas ; Bratton, Daniel J. ; Wons, Annette ; Gaisl, Thomas ; Sievi, Noriane A. ; Clarenbach, Christian F. ; Kohler, Malcolm ; Krayenbühl, Pierre A.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3550-2ab29fae0874b2fee1e2f94a8be97b08efce9607979a43c27f9c92c9afd63ded3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Adult</topic><topic>Fabry Disease - complications</topic><topic>Female</topic><topic>Humans</topic><topic>Male</topic><topic>Observational Study</topic><topic>Prevalence</topic><topic>Prospective Studies</topic><topic>Risk Factors</topic><topic>Sleep Apnea Syndromes - epidemiology</topic><topic>Sleep Apnea Syndromes - etiology</topic><topic>Sleep Apnea, Central - epidemiology</topic><topic>Sleep Apnea, Central - etiology</topic><topic>Sleep Apnea, Obstructive - epidemiology</topic><topic>Sleep Apnea, Obstructive - etiology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Franzen, Daniel</creatorcontrib><creatorcontrib>Gerard, Nicolas</creatorcontrib><creatorcontrib>Bratton, Daniel J.</creatorcontrib><creatorcontrib>Wons, Annette</creatorcontrib><creatorcontrib>Gaisl, Thomas</creatorcontrib><creatorcontrib>Sievi, Noriane A.</creatorcontrib><creatorcontrib>Clarenbach, Christian F.</creatorcontrib><creatorcontrib>Kohler, Malcolm</creatorcontrib><creatorcontrib>Krayenbühl, Pierre A.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Medicine (Baltimore)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Franzen, Daniel</au><au>Gerard, Nicolas</au><au>Bratton, Daniel J.</au><au>Wons, Annette</au><au>Gaisl, Thomas</au><au>Sievi, Noriane A.</au><au>Clarenbach, Christian F.</au><au>Kohler, Malcolm</au><au>Krayenbühl, Pierre A.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Prevalence and Risk Factors of Sleep Disordered Breathing in Fabry disease: A Prospective Cohort Study</atitle><jtitle>Medicine (Baltimore)</jtitle><addtitle>Medicine (Baltimore)</addtitle><date>2015-12-01</date><risdate>2015</risdate><volume>94</volume><issue>52</issue><spage>e2413</spage><epage>e2413</epage><pages>e2413-e2413</pages><issn>0025-7974</issn><eissn>1536-5964</eissn><abstract>Excessive daytime sleepiness (EDS) is a frequently reported and not well-understood symptom in patients with Fabry disease (FD). Sleep-disordered breathing (SDB) is a possible factor. As deposition of glycosphingolipids in the upper airway muscles is likely, we hypothesized that obstructive sleep apnoea (OSA) is highly prevalent in FD and positively associated with its severity.All patients with FD who are followed in the Fabry cohort of the University Hospital Zurich (n = 62) were asked to participate in this prospective cohort study. Eligible patients were prospectively investigated by assessing their daytime sleepiness using the Epworth Sleepiness Scale (ESS), the severity of FD using the Mainz Severity Score Index (MSSI), and by an ambulatory overnight respiratory polygraphy between November 1, 2013, and January 31, 2015. SDB was defined as an apnea/hypopnea index (AHI) of > 5/h.Fifty-two patients (mean ± SD age 42.8 ± 14.7 years, 33% men, mean ± SD BMI 23.4 ± 3.6 kg/m) with a median (IQR) MSSI of 12 (5-19) were included. Median (IQR) ESS was 6 (2-10) and 7 patients (14%) had an ESS > 10. Thirteen patients (25%) had SDB (78% obstructive sleep apnea, 22% central sleep apnea). In the multivariable analysis, the age was the only statistically significant predictor of SDB (OR 1.11, 95% CI 1.04-1.18, P = 0.001). ESS was associated with depression (P < 0.001) but not AHI nor age.This study shows that SDB, especially obstructive sleep apnea is highly prevalent in patients with Fabry disease. However, EDS in FD seems to be related with depression rather than SDB.ClinicalTrials.gov (identifier: NCT01947634).</abstract><cop>United States</cop><pub>Wolters Kluwer Health, Inc. All rights reserved</pub><pmid>26717401</pmid><doi>10.1097/MD.0000000000002413</doi><oa>free_for_read</oa></addata></record> |
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| ispartof | Medicine (Baltimore), 2015-12, Vol.94 (52), p.e2413-e2413 |
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| source | PubMed (Medline); LWW_医学期刊; IngentaConnect Journals |
| subjects | Adult Fabry Disease - complications Female Humans Male Observational Study Prevalence Prospective Studies Risk Factors Sleep Apnea Syndromes - epidemiology Sleep Apnea Syndromes - etiology Sleep Apnea, Central - epidemiology Sleep Apnea, Central - etiology Sleep Apnea, Obstructive - epidemiology Sleep Apnea, Obstructive - etiology |
| title | Prevalence and Risk Factors of Sleep Disordered Breathing in Fabry disease: A Prospective Cohort Study |
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