Cost-Utility Analysis of Continuous Positive Airway Pressure Therapy Compared With Usual Care for Obstructive Sleep Apnea in the Public Health System in Brazil.

OBJECTIVES: This study aimed to conduct a cost-utility analysis of continuous positive airway pressure (CPAP) therapy compared with usual care as treatment of moderate to severe cases of obstructive sleep apnea (OSA) in Brazil, where decentralized policies of CPAP provision are in place. METHODS: Markov cohort model comparing CPAP therapy with usual care, that is, no specific treatment for OSA, for moderate to severe cases was used. The payer perspective from the Unified Health System, Brazil, was adopted. Effectiveness parameters and costs related to health states were informed by literature review. Resource use related to CPAP therapy was defined by specialists and costs informed by recent purchase and leasing contracts. Incremental cost-effectiveness ratios were generated for purchase and leasing contracts to reflect current practices. A conservative willingness-to-pay threshold was set at 1 gross domestic product per capita per quality-adjusted life-year (QALY) (Brazilian reais [BRL] 40 712/QALY). Uncertainties were explored in deterministic and probabilistic sensitivity analyses. RESULTS: Incremental cost-effectiveness ratio for the purchase modality was 8303 BRL/QALY and for leasing 45 192 BRL/QALY. Considering the adopted willingness-to-pay threshold, provision of CPAP by the purchase modality was considered cost-effective but not the leasing modality. The parameter related to the greatest uncertainty was the reduction in the risk of having a stroke attributable to CPAP. Probabilistic analysis confirmed the robustness of results. CONCLUSIONS: CPAP therapy is a cost-effective alternative compared with usual care for moderate to severe OSA for the purchase modality. These results should help underpinning the decision making related to a uniform policy of CPAP provision across the country.

Legal action for access to resources inefficiently made available in health care systems in Brazil: a case study on obstructive sleep apnea / Judicialização para acesso a recursos disponibilizados de forma ineficiente nos sistemas de saúde do Brasil: estudo de caso sobre apneia obstrutiva do sono

ABSTRACT Objective: Obstructive sleep apnea (OSA) is a highly prevalent chronic disease, associated with morbidity and mortality. Although effective treatment for OSA is commercially available, their provision is not guaranteed by lines of care throughout Brazil, making legal action necessary. This study aimed at presenting data related to the volume of legal proceedings regarding the access to diagnosis and treatment of OSA in Brazil. Methods: This was a descriptive study of national scope, evaluating the period between January of 2016 and December of 2020. The number of lawsuits was analyzed according to the object of the demand (diagnosis or treatment). Projections of total expenses were carried out according to the number of lawsuits. Results: We identified 1,462 legal proceedings (17.6% and 82.4% related to diagnosis and treatment, respectively). The projection of expenditure for OSA diagnosis in the public and private spheres were R$575,227 and R$188,002, respectively. The projection of expenditure for OSA treatment in the public and private spheres were R$2,656,696 and R$253,050, respectively. There was a reduction in the number of lawsuits between 2017 and 2019. Conclusions: Legal action as a strategy for accessing diagnostic and therapeutic resources related to OSA is a recurrent practice, resulting in inefficiency and inequity. The reduction in the number of lawsuits between 2017 and 2019 might be explained by the expansion of local health care policies or by barriers in the journey of patients with OSA, such as difficulties in being referred to specialized health care and low availability of diagnostic resources.

RESUMO Objetivo: A apneia obstrutiva do sono (AOS) é uma doença crônica altamente prevalente, associada a morbidade e mortalidade. Embora tratamentos efetivos para a AOS estejam disponíveis comercialmente, seu fornecimento não é garantido pelos fluxos de atendimento em todo o Brasil, tornando necessária a judicialização. Este estudo teve como objetivo apresentar dados referentes ao volume de processos judiciais relacionados ao acesso ao diagnóstico e tratamento da AOS no Brasil. Métodos: Estudo descritivo de abrangência nacional, avaliando o período entre janeiro de 2016 e dezembro de 2020. O número de demandas judiciais foi analisado de acordo com o objeto da demanda (diagnóstico ou tratamento). As projeções das despesas totais foram realizadas de acordo com o número de demandas judiciais. Resultados: Foram identificados 1.462 processos judiciais (17,6% e 82,4% referentes a diagnóstico e tratamento, respectivamente). A projeção dos gastos com o diagnóstico da AOS nas esferas pública e privada foi de R$ 575.227 e R$ 188.002, respectivamente. A projeção dos gastos com o tratamento da AOS nas esferas pública e privada foi de R$ 2.656.696 e R$ 253.050, respectivamente. Houve redução do número de demandas judiciais entre 2017 e 2019. Conclusões: A judicialização como estratégia de acesso a recursos diagnósticos e terapêuticos relacionados à AOS é uma prática recorrente, resultando em ineficiência e iniquidade. A redução do número de demandas judiciais entre 2017 e 2019 pode ser explicada pela expansão das políticas locais de saúde ou por barreiras na jornada dos pacientes com AOS, como dificuldades de encaminhamento para atendimento especializado e a baixa disponibilidade de recursos diagnósticos.

Telehealth Strategies to Support Referral Management to Secondary Care in Brazil: A Cost-Effectiveness Analysis.

OBJECTIVES: This study aimed to assess the cost-effectiveness of a remotely operated referral management system (RORMS) compared with a conventional referral management system (CRMS) in Brazil. METHODS: This is a model-based cost-effectiveness analysis under the perspective of the Unified Healthcare System (Sistema Único de Saúde [SUS]) in Brazil. A Markov microsimulation model was developed to compare costs and referral outcomes of the RORMS and the CRMS. Model consisted of 4 states representative of sequential stepwise assessments of referral suitability, 3 states representative of referral outcomes, and 1 exit model state. Target population represented cases being referred from primary healthcare units to specialized care in SUS. Model inputs related to costs and effectiveness in the RORMS arm were obtained from the data set of a RORMS between July and December 2019. Model inputs for the CRMS model arm were obtained from administrative data sets of 2 Brazilian localities for the year 2019. Relative effect size of RORMS in comparison with CRMS in SUS was obtained from published studies. Effectiveness outcome was unnecessary referrals averted. The incremental cost-effectiveness ratio was calculated for the base case. Probabilistic sensitivity analysis was conducted. RESULTS: In the base-case analyses, RORMS dominated CRMS, with expected cost-savings from $50.42 to $80.62 per unnecessary referral averted. RORMS was the dominant strategy in 83.7% of 100 000 simulations in the probabilistic sensitivity analysis. In 16.2% of simulations, incremental cost-effectiveness ratio was between $0 and $222 per unnecessary referral averted. CONCLUSIONS: Model-based simulations indicate that the RORMS is likely to be cost saving in comparison with the CRMS.

Economic evaluation of CPAP therapy for obstructive sleep apnea: a scoping review and evidence map.

PURPOSE: To synthesize findings of economic evaluations investigating cost-effectiveness of continuous positive airway pressure (CPAP) for obstructive sleep apnea (OSA) and of strategies of organization of care related to CPAP therapy. METHODS: Scoping review with searches conducted in MEDLINE, CRD, LILACS, and Embase in August 2020. Eligible studies were economic evaluations comparing CPAP to other alternative or assessing strategies of care for CPAP therapy. Results were presented narratively, and incremental cost-effectiveness ratios (ICER) were presented in evidence maps. RESULTS: Of 34 studies, 3 concluded that CPAP is less costly and more effective when compared to usual care. Most studies indicated that CPAP is associated with better health outcomes, but at higher prices. ICER ranged from USD 316 to 98,793 per quality-adjusted life years (QALY) gained (median 16,499; IQR 8267 to 33,119). One study concluded that CPAP is more costly and less effective, when treatment is applied to all patients, regardless of disease severity. Variability of ICER was mainly due to definition of population and applied time horizons. When CPAP was compared to mandibular advancement device, ICER ranged from USD 21,153 to 361,028 (median 89,671; IQR 26,829 to 295,983), which represents the investment in CPAP therapy required to obtain one extra QALY. Three studies assessed the effects of organizing CPAP therapy in primary care, which was cost-effective or cost-saving. CONCLUSIONS: Compared to usual care, CPAP is cost-effective after the second year of treatment, when indicated for moderate-to-severe OSA. CPAP therapy may be even more cost-effective by using different strategies of organization of care. These findings may inform decision making related to CPAP reimbursement in health systems. CLINICAL TRIAL REGISTRATION NUMBER: Not applicable.

Telehealth to support referral management in a universal health system: a before-and-after study.

BACKGROUND: Management of patient flow within a healthcare network, allowing equitable and qualified access to healthcare, is a major challenge for universal health systems. Implementation of telehealth strategies to support referral management has been shown to increase primary care resolution and to promote coordination of care. The objective of this study was to assess the impact of telehealth strategies on waiting lists and waiting times for specialized care in Brazil. METHODS: Before-and-after study with measures obtained between January 2019 and February 2020. Baseline measurements of waiting lists were obtained immediately before the implementation of a remotely operated referral management system. Post-interventional measurements were obtained monthly, up to six months after the beginning of operation. Data was extracted from the database of the project. General linear models were applied to assess interaction of locality and time over number of cases on waiting lists and waiting times. RESULTS: At baseline, the median number of cases on waiting lists ranged from 2961 to 12,305 cases. Reductions of the number of cases on waiting lists after six months of operation were observed in all localities. The magnitude of the reduction ranged from 54.67 to 88.97 %. Interaction of time measurements was statistically significant from the second month onward. Median waiting times ranged from 159 to 241 days at baseline. After six months, there was a decrease of 100 and 114 waiting days in two localities, respectively, with reduction of waiting times only for high-risk cases in the third locality. CONCLUSIONS: Adoption of telehealth strategies resulted in the reduction of number of cases on waiting lists. Results were consistent across localities, suggesting that telehealth interventions are viable in diverse settings.

The effect of exposure to long working hours on depression: A systematic review and meta-analysis from the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury.

BACKGROUND: The World Health Organization (WHO) and the International Labour Organization (ILO) are developing the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury (WHO/ILO Joint Estimates), supported by a large number of individual experts. Evidence from previous reviews suggests that exposure to long working hours may cause depression. In this article, we present a systematic review and meta-analysis of parameters for estimating (if feasible) the number of deaths and disability-adjusted life years from depression that are attributable to exposure to long working hours, for the development of the WHO/ILO Joint Estimates. OBJECTIVES: We aimed to systematically review and meta-analyse estimates of the effect of exposure to long working hours (three categories: 41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on depression (three outcomes: prevalence, incidence and mortality). DATA SOURCES: We developed and published a protocol, applying the Navigation Guide as an organizing systematic review framework where feasible. We searched electronic academic databases for potentially relevant records from published and unpublished studies, including the WHO International Clinical Trial Registers Platform, Medline, PubMed, EMBASE, Web of Science, CISDOC and PsycInfo. We also searched grey literature databases, Internet search engines and organizational websites; hand-searched reference lists of previous systematic reviews; and consulted additional experts. STUDY ELIGIBILITY AND CRITERIA: We included working-age (≥15 years) workers in the formal and informal economy in any WHO and/or ILO Member State but excluded children (aged <15 years) and unpaid domestic workers. We included randomized controlled trials, cohort studies, case-control studies and other non-randomized intervention studies with an estimate of the effect of exposure to long working hours (41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on depression (prevalence, incidence and/or mortality). STUDY APPRAISAL AND SYNTHESIS METHODS: At least two review authors independently screened titles and abstracts against the eligibility criteria at a first stage and full texts of potentially eligible records at a second stage, followed by extraction of data from qualifying studies. Missing data were requested from principal study authors. We combined odds ratios using random-effects meta-analysis. Two or more review authors assessed the risk of bias, quality of evidence and strength of evidence, using Navigation Guide and GRADE tools and approaches adapted to this project. RESULTS: Twenty-two studies (all cohort studies) met the inclusion criteria, comprising a total of 109,906 participants (51,324 females) in 32 countries (as one study included multiple countries) in three WHO regions (Americas, Europe and Western Pacific). The exposure was measured using self-reports in all studies, and the outcome was assessed with a clinical diagnostic interview (four studies), interview questions about diagnosis and treatment of depression (three studies) or a validated self-administered rating scale (15 studies). The outcome was defined as incident depression in all 22 studies, with first time incident depression in 21 studies and recurrence of depression in one study. We did not identify any study on prevalence of depression or on mortality from depression. For the body of evidence for the outcome incident depression, we had serious concerns for risk of bias due to selection because of incomplete outcome data (most studies assessed depression only twice, at baseline and at a later follow-up measurement, and likely have missed cases of depression that occurred after baseline but were in remission at the time of the follow-up measurement) and due to missing information on life-time prevalence of depression before baseline measurement. Compared with working 35-40 h/week, we are uncertain about the effect on acquiring (or incidence of) depression of working 41-48 h/week (pooled odds ratio (OR) 1.05, 95% confidence interval (CI) 0.86 to 1.29, 8 studies, 49,392 participants, I2 46%, low quality of evidence); 49-54 h/week (OR 1.06, 95% CI 0.93 to 1.21, 8 studies, 49,392 participants, I2 40%, low quality of evidence); and ≥ 55 h/week (OR 1.08, 95% CI 0.94 to 1.24, 17 studies, 91,142 participants, I2 46%, low quality of evidence). Subgroup analyses found no evidence for statistically significant (P < 0.05) differences by WHO region, sex, age group and socioeconomic status. Sensitivity analyses found no statistically significant differences by outcome measurement (clinical diagnostic interview [gold standard] versus other measures) and risk of bias ("high"/"probably high" ratings in any domain versus "low"/"probably low" in all domains). CONCLUSIONS: We judged the existing bodies of evidence from human data as "inadequate evidence for harmfulness" for all three exposure categories, 41-48, 48-54 and ≥55 h/week, for depression prevalence, incidence and mortality; the available evidence is insufficient to assess effects of the exposure. Producing estimates of the burden of depression attributable to exposure to long working appears not evidence-based at this point. Instead, studies examining the association between long working hours and risk of depression are needed that address the limitations of the current evidence.

The effect of exposure to long working hours on alcohol consumption, risky drinking and alcohol use disorder: A systematic review and meta-analysis from the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury.

BACKGROUND: The World Health Organization (WHO) and the International Labour Organization (ILO) are developing Joint Estimates of the work-related burden of disease and injury (WHO/ILO Joint Estimates), with contributions from a large network of experts. Evidence from mechanistic data suggests that exposure to long working hours may increase alcohol consumption and cause alcohol use disorder. In this paper, we present a systematic review and meta-analysis of parameters for estimating the number of deaths and disability-adjusted life years from alcohol consumption and alcohol use disorder that are attributable to exposure to long working hours, for the development of the WHO/ILO Joint Estimates. OBJECTIVES: We aimed to systematically review and meta-analyse estimates of the effect of exposure to long working hours (three categories: 41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on alcohol consumption, risky drinking (three outcomes: prevalence, incidence and mortality) and alcohol use disorder (three outcomes: prevalence, incidence and mortality). DATA SOURCES: We developed and published a protocol, applying the Navigation Guide as an organizing systematic review framework where feasible. We searched electronic bibliographic databases for potentially relevant records from published and unpublished studies, including the WHO International Clinical Trials Register, Ovid MEDLINE, PubMed, Embase, and CISDOC on 30 June 2018. Searches on PubMed were updated on 18 April 2020. We also searched electronic grey literature databases, Internet search engines and organizational websites; hand-searched reference list of previous systematic reviews and included study records; and consulted additional experts. STUDY ELIGIBILITY AND CRITERIA: We included working-age (≥15 years) workers in the formal and informal economy in any WHO and/or ILO Member State but excluded children (<15 years) and unpaid domestic workers. We considered for inclusion randomized controlled trials, cohort studies, case-control studies and other non-randomized intervention studies with an estimate of the effect of exposure to long working hours (41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on alcohol consumption (in g/week), risky drinking, and alcohol use disorder (prevalence, incidence or mortality). STUDY APPRAISAL AND SYNTHESIS METHODS: At least two review authors independently screened titles and abstracts against the eligibility criteria at a first stage and full texts of potentially eligible records at a second stage, followed by extraction of data from publications related to qualifying studies. Two or more review authors assessed the risk of bias, quality of evidence and strength of evidence, using Navigation Guide and GRADE tools and approaches adapted to this project. RESULTS: Fourteen cohort studies met the inclusion criteria, comprising a total of 104,599 participants (52,107 females) in six countries of three WHO regions (Americas, South-East Asia, and Europe). The exposure and outcome were assessed with self-reported measures in most studies. Across included studies, risk of bias was generally probably high, with risk judged high or probably high for detection bias and missing data for alcohol consumption and risky drinking. Compared to working 35-40 h/week, exposure to working 41-48 h/week increased alcohol consumption by 10.4 g/week (95% confidence interval (CI) 5.59-15.20; seven studies; 25,904 participants, I2 71%, low quality evidence). Exposure to working 49-54 h/week increased alcohol consumption by 17.69 g/week (95% confidence interval (CI) 9.16-26.22; seven studies, 19,158 participants, I2 82%, low quality evidence). Exposure to working ≥55 h/week increased alcohol consumption by 16.29 g/week (95% confidence interval (CI) 7.93-24.65; seven studies; 19,692 participants; I2 82%, low quality evidence). We are uncertain about the effect of exposure to working 41-48 h/week, compared with working 35-40 h/week on developing risky drinking (relative risk 1.08; 95% CI 0.86-1.36; 12 studies; I2 52%, low certainty evidence). Working 49-54 h/week did not increase the risk of developing risky drinking (relative risk 1.12; 95% CI 0.90-1.39; 12 studies; 3832 participants; I2 24%, moderate certainty evidence), nor working ≥55 h/week (relative risk 1.11; 95% CI 0.95-1.30; 12 studies; 4525 participants; I2 0%, moderate certainty evidence). Subgroup analyses indicated that age may influence the association between long working hours and both alcohol consumption and risky drinking. We did not identify studies for which we had access to results on alcohol use disorder. CONCLUSIONS: Overall, for alcohol consumption in g/week and for risky drinking, we judged this body of evidence to be of low certainty. Exposure to long working hours may have increased alcohol consumption, but we are uncertain about the effect on risky drinking. We found no eligible studies on the effect on alcohol use disorder. Producing estimates for the burden of alcohol use disorder attributable to exposure to long working hours appears to not be evidence-based at this time. PROTOCOL IDENTIFIER: https://doi.org/10.1016/j.envint.2018.07.025. PROSPERO REGISTRATION NUMBER: CRD42018084077.

The effect of exposure to long working hours on ischaemic heart disease: A systematic review and meta-analysis from the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury.

BACKGROUND: The World Health Organization (WHO) and the International Labour Organization (ILO) are developing Joint Estimates of the work-related burden of disease and injury (WHO/ILO Joint Estimates), with contributions from a large network of experts. Evidence from mechanistic data suggests that exposure to long working hours may cause ischaemic heart disease (IHD). In this paper, we present a systematic review and meta-analysis of parameters for estimating the number of deaths and disability-adjusted life years from IHD that are attributable to exposure to long working hours, for the development of the WHO/ILO Joint Estimates. OBJECTIVES: We aimed to systematically review and meta-analyse estimates of the effect of exposure to long working hours (three categories: 41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on IHD (three outcomes: prevalence, incidence and mortality). DATA SOURCES: We developed and published a protocol, applying the Navigation Guide as an organizing systematic review framework where feasible. We searched electronic databases for potentially relevant records from published and unpublished studies, including MEDLINE, Scopus, Web of Science, CISDOC, PsycINFO, and WHO ICTRP. We also searched grey literature databases, Internet search engines and organizational websites; hand-searched reference lists of previous systematic reviews; and consulted additional experts. STUDY ELIGIBILITY AND CRITERIA: We included working-age (≥15 years) workers in the formal and informal economy in any WHO and/or ILO Member State but excluded children (aged < 15 years) and unpaid domestic workers. We included randomized controlled trials, cohort studies, case-control studies and other non-randomized intervention studies which contained an estimate of the effect of exposure to long working hours (41-48, 49-54 and ≥55 h/week), compared with exposure to standard working hours (35-40 h/week), on IHD (prevalence, incidence or mortality). STUDY APPRAISAL AND SYNTHESIS METHODS: At least two review authors independently screened titles and abstracts against the eligibility criteria at a first stage and full texts of potentially eligible records at a second stage, followed by extraction of data from qualifying studies. Missing data were requested from principal study authors. We combined relative risks using random-effect meta-analysis. Two or more review authors assessed the risk of bias, quality of evidence and strength of evidence, using Navigation Guide and GRADE tools and approaches adapted to this project. RESULTS: Thirty-seven studies (26 prospective cohort studies and 11 case-control studies) met the inclusion criteria, comprising a total of 768,751 participants (310,954 females) in 13 countries in three WHO regions (Americas, Europe and Western Pacific). The exposure was measured using self-reports in all studies, and the outcome was assessed with administrative health records (30 studies) or self-reported physician diagnosis (7 studies). The outcome was defined as incident non-fatal IHD event in 19 studies (8 cohort studies, 11 case-control studies), incident fatal IHD event in two studies (both cohort studies), and incident non-fatal or fatal ("mixed") event in 16 studies (all cohort studies). Because we judged cohort studies to have a relatively lower risk of bias, we prioritized evidence from these studies and treated evidence from case-control studies as supporting evidence. For the bodies of evidence for both outcomes with any eligible studies (i.e. IHD incidence and mortality), we did not have serious concerns for risk of bias (at least for the cohort studies). No eligible study was found on the effect of long working hours on IHD prevalence. Compared with working 35-40 h/week, we are uncertain about the effect on acquiring (or incidence of) IHD of working 41-48 h/week (relative risk (RR) 0.98, 95% confidence interval (CI) 0.91 to 1.07, 20 studies, 312,209 participants, I2 0%, low quality of evidence) and 49-54 h/week (RR 1.05, 95% CI 0.94 to 1.17, 18 studies, 308,405 participants, I2 0%, low quality of evidence). Compared with working 35-40 h/week, working ≥55 h/week may have led to a moderately, clinically meaningful increase in the risk of acquiring IHD, when followed up between one year and 20 years (RR 1.13, 95% CI 1.02 to 1.26, 22 studies, 339,680 participants, I2 5%, moderate quality of evidence). Compared with working 35-40 h/week, we are very uncertain about the effect on dying (mortality) from IHD of working 41-48 h/week (RR 0.99, 95% CI 0.88 to 1.12, 13 studies, 288,278 participants, I2 8%, low quality of evidence) and 49-54 h/week (RR 1.01, 95% CI 0.82 to 1.25, 11 studies, 284,474 participants, I2 13%, low quality of evidence). Compared with working 35-40 h/week, working ≥55 h/week may have led to a moderate, clinically meaningful increase in the risk of dying from IHD when followed up between eight and 30 years (RR 1.17, 95% CI 1.05 to 1.31, 16 studies, 726,803 participants, I2 0%, moderate quality of evidence). Subgroup analyses found no evidence for differences by WHO region and sex, but RRs were higher among persons with lower SES. Sensitivity analyses found no differences by outcome definition (exclusively non-fatal or fatal versus "mixed"), outcome measurement (health records versus self-reports) and risk of bias ("high"/"probably high" ratings in any domain versus "low"/"probably low" in all domains). CONCLUSIONS: We judged the existing bodies of evidence for human evidence as "inadequate evidence for harmfulness" for the exposure categories 41-48 and 49-54 h/week for IHD prevalence, incidence and mortality, and for the exposure category ≥55 h/week for IHD prevalence. Evidence on exposure to working ≥55 h/week was judged as "sufficient evidence of harmfulness" for IHD incidence and mortality. Producing estimates for the burden of IHD attributable to exposure to working ≥55 h/week appears evidence-based, and the pooled effect estimates presented in this systematic review could be used as input data for the WHO/ILO Joint Estimates.