Feasibility and Long-Term Compliance to Continuous Positive Airway Pressure Treatment in Adults With Down Syndrome, a Genetic Form of Alzheimer's Disease

Sandra Giménez; Ariadna Farre; Fátima Morente; Laura Videla; Marta Gutiérrez; Susana Clos; Ana Fernández; Marta Blanco; Miren Altuna; Jordi Pegueroles; Amparo Asensio; Bessy Benejam; Mar Batista; Isabel Barroeta; Ana Fortuna; Juan Fortea; Mercedes Mayos

doi:10.3389/fnins.2022.838412

Feasibility and Long-Term Compliance to Continuous Positive Airway Pressure Treatment in Adults With Down Syndrome, a Genetic Form of Alzheimer's Disease

Front Neurosci. 2022 Mar 30:16:838412. doi: 10.3389/fnins.2022.838412. eCollection 2022.

Authors

Sandra Giménez¹, Ariadna Farre¹, Fátima Morente¹, Laura Videla^{2

3

4}, Marta Gutiérrez¹, Susana Clos², Ana Fernández¹, Marta Blanco¹, Miren Altuna^{2

3}, Jordi Pegueroles², Amparo Asensio¹, Bessy Benejam^{2

3

4}, Mar Batista¹, Isabel Barroeta^{2

3}, Ana Fortuna¹, Juan Fortea^{2

3

4}, Mercedes Mayos^{1

5}

Affiliations

¹ Multidisciplinary Sleep Unit, Respiratory Department, Hospital de la Santa Creu i Sant Pau, Biomedical Research Institute Sant Pau, Barcelona, Spain.
² Sant Pau Memory Unit, Department of Neurology, Hospital de la Santa Creu i Sant Pau, Biomedical Research Institute Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain.
³ Center of Biomedical Investigation Network for Neurodegenerative Diseases (CIBERNED), Madrid, Spain.
⁴ Barcelona Down Medical Center, Fundació Catalana de Síndrome de Down, Barcelona, Spain.
⁵ Centro de Investigación Biomédica en Red de Enfermedades Respiratorias (CIBERNED), Madrid, Spain.

Abstract

Background: Down syndrome (DS) is a genetic form of Alzheimer's disease (AD) with a high prevalence of obstructive sleep apnea (OSA). These characteristics place the DS population as an optimal model to study the relationship between sleep and AD and to design clinical trials of preventive sleep therapies for AD. Regrettably, OSA treatment with continuous positive airway pressure (CPAP) is often neglected in adults with DS. In both clinical practice and research trials, it is usually presumed that these patients will not adapt to or tolerate the therapy.

Study objective: We aimed to evaluate the feasibility and long-term CPAP compliance in this population and their capacity to be enrolled in CPAP research studies.

Methods: We prospectively compared the CPAP compliance of 17 OSA patients with DS and 19 age and sex matched OSA euploid patients. CPAP management and follow-up schedules were prescribed according to the habitual clinical practice. We compared group differences in tolerance, objective, and subjective hours of nightly CPAP usage at the 1st, 3rd, 6th, 12th, 24th, and 36th month visits. Good compliance was defined as at least 4 h use per night. We also investigated predictive factors of long-term CPAP compliance.

Results: The percentage of DS subjects with good CPAP compliance (81.2 vs. 78.9%) and the objective CPAP use (5 vs. 6 h, p = 0.92) did not differ from the control group (CG). Subjective CPAP compliance was significantly higher in OSA patients with DS than in controls in all the follow-up visits (8 vs. 6.75 h, p = 0.001). The DS group had a significantly higher number of visits (9 vs. 5; p = 0.021) and mask changes (2.5 vs. 2; p = 0.05) than controls. Objective hours of CPAP use at the first follow-up visit predicted long-term CPAP compliance (p < 0.005).

Conclusion: CPAP treatment is feasible and has good long-term compliance in OSA patients with DS. It should be recommended to improve health and prevent comorbidities. The DS population is indeed suitable to participate in longitudinal preventive sleep clinical trials for AD.

Keywords: Alzheimer’s disease; CPAP compliance; Down syndrome; obstructive sleep apnea; sleep.