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Purpose <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.</p> Methods <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 3–9 months post-CPAP were analyzed. The Epworth Sleepiness Scale (ESS) was used to assess daytime sleepiness.</p> Results <p>68 cases (47% hEDS/53% HSD, Beighton score 5.71 ± 2.29, age 34.07 ± 13.28, BMI 32.88 ± 8.47) were matched to 68 controls. All pairs completed PSG; 53 pairs pursued CPAP; 41 pairs were reevaluated. Cases had lower sleep efficiency (77%±16% vs. 82%±10%, <i>p</i> &lt; 0.05) and higher insomnia prevalence (44.7% vs. 34.3%, <i>p</i> &lt; 0.05). Pre-CPAP ESS was similar (10.39 ± 4.89 vs. 11.22 ± 5.12, <i>p</i> &gt; 0.05). CPAP reduced ESS in controls (− 4.46, <i>p</i> &lt; 0.05) but not in cases (− 1.56, <i>p</i> &gt; 0.05). Cases and controls had similar CPAP compliance (75.6%±25.2% vs. 74.6%±26.7%, <i>p</i> &gt; 0.05), Apnea-Hypopnea Index (AHI) (7.2 [13.6] vs. 7.0 [12.7], <i>p</i> &gt; 0.05), lowest SpO<sub>2</sub> (86.08%<b>±</b>4.31% vs. 85.67%<b>±</b>3.52%, <i>p</i> &gt; 0.05), and residual AHI (1.55 [1.75] vs. 1.2 [1.35], <i>p</i> &gt; 0.05). Cases used nasal masks less frequently (22.6% vs. 41.5%, <i>p</i> &lt; 0.05).</p> Conclusion <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.</p>

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Obstructive sleep apnea and CPAP efficacy in patients with Hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorder: a case-control study

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年7月8日

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  • Kenneth L. Zhang,
  • Rebecca Wallon,
  • Christina M. Laukaitis,
  • Charles Davies

摘要

Purpose

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.

Methods

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 3–9 months post-CPAP were analyzed. The Epworth Sleepiness Scale (ESS) was used to assess daytime sleepiness.

Results

68 cases (47% hEDS/53% HSD, Beighton score 5.71 ± 2.29, age 34.07 ± 13.28, BMI 32.88 ± 8.47) were matched to 68 controls. All pairs completed PSG; 53 pairs pursued CPAP; 41 pairs were reevaluated. Cases had lower sleep efficiency (77%±16% vs. 82%±10%, p < 0.05) and higher insomnia prevalence (44.7% vs. 34.3%, p < 0.05). Pre-CPAP ESS was similar (10.39 ± 4.89 vs. 11.22 ± 5.12, p > 0.05). CPAP reduced ESS in controls (− 4.46, p < 0.05) but not in cases (− 1.56, p > 0.05). Cases and controls had similar CPAP compliance (75.6%±25.2% vs. 74.6%±26.7%, p > 0.05), Apnea-Hypopnea Index (AHI) (7.2 [13.6] vs. 7.0 [12.7], p > 0.05), lowest SpO2 (86.08%±4.31% vs. 85.67%±3.52%, p > 0.05), and residual AHI (1.55 [1.75] vs. 1.2 [1.35], p > 0.05). Cases used nasal masks less frequently (22.6% vs. 41.5%, p < 0.05).

Conclusion

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.