Original Title: Terapia miofuncional (ejercicios orofaríngeos) para la apnea obstructiva del sueńo

Authors' objectives: • To evaluate the effectiveness and safety of myofunctional therapy for OSA treatment. • To review the available literature on the cost-effectiveness of myofunctional therapy for OSA. • To assess the costs of this therapy compared to standard clinical practice in Spain from the perspective of the National Health System (NHS). • To identify ethical, legal, organizational, and social considerations associated with myofunctional therapy. • To pinpoint research needs and standard outcome measures from the perspectives of patients, caregivers, healthcare professionals, and researchers.

Authors' results and conclusions: RESULTS EFFECTIVENESS AND SAFETY In the preliminary search, a high-quality systematic review (SR) addressing the effectiveness and safety of myofunctional therapy for treating OSA was identified, covering all available evidence up to May 2020. Subsequently, nine additional publications were identified. Ultimately, a total of 15 randomized controlled trials (RCTs) (18 references) involving 597 participants were included in the report. The mean age of participants was 50.53 ± 14.34 years, with only one study focusing on a pediatric population, and 67.57% of participants were men. MYOFUNCTIONAL THERAPY VS. NO INTERVENTION, WAITLIST, OR PLACEBO/SHAM Ten RCTs assessed the effectiveness of myofunctional therapy compared to no intervention, waitlist, or placebo/sham. No significant differences were observed in health-related quality of life (HRQoL) (3 studies, N = 56, MD = 1.2, 95% CI: -1.2 to 3.61, very low evidence quality), sleep efficiency (3 studies, N = 108, MD = 0.78%, 95% CI: -3.13 to 4.68, very low evidence quality), oxygen desaturation index (3 studies, N = 83, MD = -4.73, 95% CI: -14.11 to 4.65, very low evidence quality), or average oxygen saturation (2 studies, N = 79, MD = -1.18, 95% CI: -0.68 to 3.04, very low evidence quality). However, significant improvements were observed in sleep quality (6 studies, N = 164, MD = -2.03, 95% CI: -2.86 to -1.19, very low evidence quality), daytime sleepiness (8 studies, N = 235, SMD = -0.85, 95% CI: -1.12 to -0.57, low evidence quality), minimum oxygen saturation (5 studies, N = 174, MD = 2.91%, 95% CI: 1.27 to 4.55, low evidence quality), and apnea-hypopnea index (AHI) (8 studies, N = 232, MD = -5.55 events/hour, 95% CI: -9.53 to -1.57, low evidence quality). MYOFUNCTIONAL THERAPY VS. CPAP A single RCT evaluated the effectiveness of myofunctional therapy compared to CPAP. No significant differences were observed in HRQoL (low evidence quality), daytime sleepiness (MD = 0.30, 95% CI: -1.65 to 2.25, low evidence quality), or sleep efficiency (MD = -2.10, 95% CI: -7.85 to 3.65, low evidence quality). Regarding AHI, the CPAP group achieved significantly lower values than the myofunctional therapy group (MD = 9.60, 95% CI: 2.46 to 16.74, low evidence quality). Additionally, minimum and average oxygen saturation levels were significantly lower in participants who received myofunctional therapy compared to those treated with CPAP (MD = -5.3, 95% CI: -8.89 to -1.71, and MD = -1.4, 95% CI: -2.12 to -0.68, respectively). MYOFUNCTIONAL THERAPY COMBINED WITH CPAP VS. CPAP ALONE Three RCTs evaluated the effectiveness of adding myofunctional therapy to CPAP treatment compared to CPAP alone. Results showed that the combination significantly improved CPAP adherence, increasing average nightly use (2 studies, N = 119, MD = 0.87 hours, 95% CI: 0.33 to 1.41, low evidence quality). However, no significant differences were observed in critical outcomes such as sleep quality (1 study, MD = -0.30, 95% CI: -2.81 to 2.21, low evidence quality) or daytime sleepiness (3 studies, N = 119, MD = -1.10, 95% CI: -3.52 to 1.32, low evidence quality). HRQoL results were mixed. Additionally, no differences were observed in AHI (2 studies, N = 119, MD = -1.06, 95% CI: -2.86 to 0.75, low evidence quality) or minimum oxygen saturation (2 studies, N = 119, MD = -0.82, 95% CI: -2.78 to 1.14, low evidence quality). SAFETY OF MYOFUNCTIONAL THERAPY Only four studies reported data on adverse events as defined by the respective studies. Overall, no serious adverse events were reported. COST-EFFECTIVENESS The SR of economic evaluations did not identify any studies meeting the established inclusion criteria. The cost analysis estimated that incorporating myofunctional therapy into the NHS would result in an incremental cost of €279.08 per patient. The sensitivity analysis revealed a cost variation ranging from €60.6 to €717.96 per patient when adjusting session unit costs and from €46.51 to €1,395.38 per patient when modifying treatment characteristics. ETHICAL, LEGAL, ORGANIZATIONAL, AND SOCIAL ASPECTS The scoping phase did not identify relevant studies on the ethical, legal, social, or organizational aspects of the evaluated technology. Consultations with three clinical experts in speech therapy and pulmonology highlighted key barriers to implementing myofunctional therapy for OSA treatment. Challenges included lack of awareness about the technique, limited specialized training, and a shortage of adequately trained professionals. Nevertheless, the experts deemed implementation feasible with appropriate resources, citing the therapy's low cost and technical simplicity, which facilitate its integration into the NHS. However, equitable access could be compromised by unequal distribution of professionals and specialized centers, emphasizing the need for investment in training and accessible healthcare infrastructure. The therapy was well-received for its non-invasive nature, simplicity, and ease of clinical integration, with strong acceptance from both patients and healthcare professionals. Despite its potential, current limitations in the availability of speech therapists within the NHS constrain its feasibility. To maximize its positive impact, professional training, robust scientific evidence, and specific training programs must be prioritized. RESEARCH NEEDS AND STANDARD OUTCOME MEASURES The report identified key research needs and necessary outcome measures for evaluating the effectiveness of myofunctional therapy in OSA, incorporating the perspectives of patients, professionals, researchers, and healthcare managers. These needs include improving future study designs, such as adequately powered RCTs, long-term follow-ups, and subgroup analyses by severity, gender, and age. Cost-effectiveness studies, qualitative research on experiences and barriers, and organizational evaluations for integrating the therapy into the NHS are also needed. Additionally, standardized protocols for myofunctional therapy, its combination with other interventions like CPAP, and personalized exercises based on individual patient characteristics should be developed. Through the James Lind Alliance (JLA) initiative, research priorities were identified concerning improved access, service quality, and the use of complementary therapies for OSA. However, no specific publications on relevant standard outcome measures were found in sources like COMET or ICHOM. CONCLUSIONS • Myofunctional therapy for OSA patients, compared to no intervention, waitlist, or placebo/sham, demonstrated benefits in outcomes such as sleep quality, daytime sleepiness, reduction in the apnea-hypopnea index (AHI), and minimum oxygen saturation during sleep. However, no significant improvements were observed in other critical outcomes, such as sleep efficiency, oxygen desaturation index (ODI), or sleep-specific quality of life. The evidence quality for most outcomes was rated very low, primarily due to high risk of bias, study heterogeneity, and imprecise confidence intervals. • Compared to CPAP, myofunctional therapy was not superior in any evaluated outcomes. CPAP was significantly more effective in reducing AHI. However, myofunctional therapy showed a significant improvement in the physical dimension of quality of life (HRQoL) in a single study. The evidence quality for this comparison was also rated very low. • The combination of myofunctional therapy and CPAP, compared to CPAP alone, significantly improved CPAP adherence, increasing average nightly use. However, no additional benefits were observed in other key outcomes, such as AHI or HRQoL. The evidence quality for this comparison was rated very low. • Myofunctional therapy was considered a safe intervention. None of the included studies reported serious adverse events, except for one isolated case of intraoral mucosal abrasion, which resolved after temporarily halting treatment. • Myofunctional therapy may be considered a complementary option for OSA patients with mild or moderate severity who do not tolerate CPAP or seek non-invasive alternatives. However, its effectiveness is limited compared to CPAP, and it is not recommended as a standalone standard treatment. • No economic evaluations meeting the inclusion criteria were identified for myofunctional therapy in OSA patients. • The cost analysis conducted in this report indicated that including myofunctional therapy as a complementary treatment to standard care (CPAP) would result in an incremental cost of €279.08 per patient from the perspective of the national health system (NHS). This cost could vary significantly depending on session frequency and treatment duration, underscoring the need for standardized protocols to optimize implementation. • No relevant studies on the ethical, legal, social, or organizational aspects of the evaluated technology were identified in the scoping phase. However, consultations with experts highlighted key barriers, such as a lack of awareness about the technique, insufficient specialized training, and a shortage of adequately trained professionals. Despite these challenges, experts considered its implementation feasible with appropriate resources, given its low cost and technical simplicity, although inequities in access due to uneven distribution of professionals and specialized centers could arise. The therapy is well accepted for its non-invasive nature and ease of clinical integration, with high receptivity among patients and professionals. • Further studies with robust methodologies, large samples, and prolonged follow-ups are needed to evaluate the effectiveness and safety of myofunctional therapy in OSA patients, as well as to collect data on resource use and costs to inform its cost-effectiveness. Additionally, qualitative studies exploring patients’ and professionals’ perceptions, experiences, and implementation barriers are necessary. Future research should also address aspects such as improving accessibility, service coordination, and quality in public health services for OSA patients, combining myofunctional therapy with CPAP, and identifying alternative effective treatments.

Authors' recommendations: MYOFUNCTIONAL THERAPY VS. NO INTERVENTION, WAITLIST, OR PLACEBO/SHAM Based on the findings of this report, a conditional recommendation is issued in favor of myofunctional therapy for individuals with OSA who do not tolerate CPAP or seek less invasive alternatives. This recommendation is based on the low certainty of the technology’s effectiveness and the lack of evidence on its associated costs. MYOFUNCTIONAL THERAPY VS. CPAP Based on the findings of this report, a weak recommendation is issued against fully substituting CPAP with myofunctional therapy for adults with OSA in the healthcare system. This recommendation is based on the lack of evidence supporting the superiority of myofunctional therapy over CPAP for key outcomes. While myofunctional therapy might be an alternative for individuals who reject CPAP, it is not recommended as a first-line treatment for all OSA patients. MYOFUNCTIONAL THERAPY COMBINED WITH CPAP VS. CPAP ALONE Based on the findings of this report, a conditional recommendation is issued in favor of including myofunctional therapy alongside CPAP for adults with OSA in the healthcare system. This recommendation is based on the low certainty of the technology’s effectiveness and the increased costs associated with its implementation compared to standard clinical practice. While myofunctional therapy appears to be a promising alternative that enhances CPAP adherence, further high-quality, low-bias research is required to increase confidence in its effectiveness and safety and to evaluate its economic impact in real-world settings.

Authors' methods: EFFECTIVENESS AND SAFETY A systematic review (SR) was conducted to evaluate the effectiveness and safety of myofunctional therapy in patients with OSA. Initially, a preliminary search was performed to identify previous SRs or Health Technology Assessment (HTA) reports published or underway by other agencies. These reports were considered for update, adaptation, or adoption based on their scope and methodological rigor. Subsequently, primary studies were identified, assessed, and synthesized. Comprehensive searches were conducted in electronic databases, including MEDLINE, Embase, CINAHL, CENTRAL, PEDro, and additional sources such as international clinical trial registries. Search strategies combined terms related to the population, intervention, and study design, with no language restrictions. Randomized controlled trials (RCTs) involving patients diagnosed with OSA and evaluating myofunctional therapy—alone or combined with other techniques—against no intervention, waitlist, placebo/sham, or active interventions were included. Study selection was performed independently by two reviewers, resolving discrepancies through consensus or involvement of a third reviewer. The methodological quality of the identified SRs was assessed using the AMSTAR-2 scale, while the risk of bias in the studies was evaluated using the RoB 2.0 tool. Meta-analyses were conducted when studies were homogeneous in terms of population, intervention, and outcomes. RevMan software was used to estimate intervention effects through mean differences or standardized mean differences. The quality of evidence and the strength of recommendations were assessed following the methodology of the international Grading of Recommendations Assessment, Development and Evaluation (GRADE) working group.   COST-EFFECTIVENESS An SR of economic evaluations of myofunctional therapy for OSA treatment was conducted. Economic evaluations (alongside primary studies or model-based) reporting one or more of the following outcomes were included: incremental cost-effectiveness ratio (ICER), costs in monetary units, and benefits expressed as quality-adjusted life years (QALYs), life years gained (LYG), monetary units, or other measures of effectiveness outcomes. The methodological quality of the studies was evaluated using Drummond’s criteria, and data were extracted and synthesized narratively, including tabulated results. COST ANALYSIS A cost analysis was conducted to estimate the incremental cost of implementing myofunctional therapy for OSA treatment within the NHS, focusing exclusively on direct healthcare costs (e.g., session costs). The base case evaluated a treatment regimen (complementary to standard clinical practice—CPAP) consisting of weekly sessions with a speech therapist or physiotherapist over 12 weeks. Additionally, a deterministic sensitivity analysis was performed to assess variations in session costs and treatment characteristics (frequency and duration of sessions). ETHICAL, LEGAL, ORGANIZATIONAL, AND SOCIAL ASPECTS These aspects were analyzed using an algorithm to evaluate the ethical, legal, organizational, social, and environmental dimensions specific to the technology. The algorithm is based on scoping to identify any relevant uncertainties for decision-making about these aspects and to explore the intervention's context. Relevant uncertainties were defined as clear research questions specific to the evaluated technology that should be addressed to adequately assess these aspects. A preliminary search was conducted in PubMed, CINAHL, and Google Scholar using various combinations of keywords derived from the search strategy used in the effectiveness section and terms specific to these dimensions. The scoping did not reveal significant uncertainties that could be addressed through an SR or additional primary study. However, uncertainties were identified regarding the technology’s applicability in the NHS, particularly in terms of equity of access, as well as technical and organizational feasibility. To contextualize the use of the technology within the healthcare system, semi-structured consultations were conducted through online meetings with clinical experts. IDENTIFICATION OF RESEARCH NEEDS AND STANDARD OUTCOME MEASURES Uncertainty areas and necessary outcome measures for evaluating the effectiveness of myofunctional therapy in OSA were identified, considering the perspectives of patients, professionals, researchers, and healthcare managers. Research needs identified in the SRs conducted for other sections of this report were analyzed alongside specialized sources, such as the James Lind Alliance (JLA), Core Outcome Measures in Effectiveness Trials (COMET), and the International Consortium for Health Outcomes Measurement (ICHOM). Study selection included publications in English and Spanish, excluding conference abstracts and texts in other languages. Data were extracted by one reviewer and validated by a second in cases of doubt. Findings were synthesized narratively, highlighting the primary research needs and key measures required to enhance the comparison and evaluation of future interventions.

Project Status: Completed

Year Published: 2024

URL for published report: https://sescs.es/en/myofunctional-therapy-osa/

English language abstract: An English language summary is available

Publication Type: Full HTA

Country: Spain

Organisation Name: Canary Health Service

Contact Address: Dirección del Servicio. Servicio Canario de la Salud, Camino Candelaria 44, 1ª planta, 38109 El Rosario, Santa Cruz de Tenerife

Contact Name: sescs@sescs.es

Contact Email: sescs@sescs.es