{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T13:27:57Z","timestamp":1787146077004,"version":"build-2736575974"},"reference-count":29,"publisher":"Springer Science and Business Media LLC","issue":"4","license":[{"start":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T00:00:00Z","timestamp":1783468800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T00:00:00Z","timestamp":1783468800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Sleep Breath"],"published-print":{"date-parts":[[2026,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Purpose<\/jats:title>\n                    <jats:p>Hypermobile Ehlers-Danlos syndrome (hEDS) and Hypermobility Spectrum Disorder (HSD) are hereditary tissue disorders existing on a spectrum associated with insomnia and obstructive sleep apnea (OSA). Oropharyngeal abnormalities in hEDS\/HSD may exacerbate OSA; this study investigates if continuous positive airway pressure (CPAP) adequately decreases daytime sleepiness in hEDS\/HSD patients with OSA.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A single institution retrospective review was conducted from 2020 to 2024. Adults with hEDS\/HSD and OSA (cases) were matched based on sex, age, race, and body mass index (BMI) to adults with OSA (controls). Data from polysomnogram (PSG), sleep clinic consults, and reevaluation 3\u20139 months post-CPAP were analyzed. The Epworth Sleepiness Scale (ESS) was used to assess daytime sleepiness.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      68 cases (47% hEDS\/53% HSD, Beighton score 5.71\u2009\u00b1\u20092.29, age 34.07\u2009\u00b1\u200913.28, BMI 32.88\u2009\u00b1\u20098.47) were matched to 68 controls. All pairs completed PSG; 53 pairs pursued CPAP; 41 pairs were reevaluated. Cases had lower sleep efficiency (77%\u00b116% vs. 82%\u00b110%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05) and higher insomnia prevalence (44.7% vs. 34.3%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05). Pre-CPAP ESS was similar (10.39\u2009\u00b1\u20094.89 vs. 11.22\u2009\u00b1\u20095.12,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05). CPAP reduced ESS in controls (\u2212\u20094.46,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05) but not in cases (\u2212\u20091.56,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05). Cases and controls had similar CPAP compliance (75.6%\u00b125.2% vs. 74.6%\u00b126.7%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05), Apnea-Hypopnea Index (AHI) (7.2 [13.6] vs. 7.0 [12.7],\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05), lowest SpO\n                      <jats:sub>2<\/jats:sub>\n                      (86.08%\n                      <jats:bold>\u00b1<\/jats:bold>\n                      4.31% vs. 85.67%\n                      <jats:bold>\u00b1<\/jats:bold>\n                      3.52%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05), and residual AHI (1.55 [1.75] vs. 1.2 [1.35],\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&gt;\u20090.05). Cases used nasal masks less frequently (22.6% vs. 41.5%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>CPAP was not associated with a statistically significant improvement in daytime sleepiness in hEDS\/HSD patients despite effective treatment, suggesting that hypermobility-related factors beyond OSA may contribute to residual sleepiness.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1007\/s11325-026-03763-3","type":"journal-article","created":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T11:47:02Z","timestamp":1783511222000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Obstructive sleep apnea and CPAP efficacy in patients with Hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorder: a case-control study"],"prefix":"10.1007","volume":"30","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7769-5611","authenticated-orcid":false,"given":"Kenneth L.","family":"Zhang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-1053-0645","authenticated-orcid":false,"given":"Rebecca","family":"Wallon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1466-7941","authenticated-orcid":false,"given":"Christina M.","family":"Laukaitis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4427-8683","authenticated-orcid":false,"given":"Charles","family":"Davies","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,7,8]]},"reference":[{"key":"3763_CR1","doi-asserted-by":"publisher","first-page":"8","DOI":"10.1002\/ajmg.c.31552","volume":"175","author":"F Malfait","year":"2017","unstructured":"Malfait F, Francomano C, Byers P et al (2017) The 2017 international classification of the Ehlers-Danlos syndromes. 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The Human Investigation Committee (IRB) of Carle Foundation Hospital approved this study (project No. 24NPR3999).","order":1,"name":"Ethics","label":"Ethics approval","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Waived by IRB.","order":2,"name":"Ethics","label":"Consent to participate","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Waived by IRB.","order":3,"name":"Ethics","label":"Consent for publication","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"All authors certify that they have no financial or non-financial interests in the subject matter or materials discussed in this manuscript.","order":4,"name":"Ethics","label":"Competing interest","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"212"}}