Epidemiology, patient journey and unmet needs related to hemophilia in Brazil: a scoping review with evidence map.

INTRODUCTION: Brazil is one of the countries with the largest population of people with hemophilia (PwH) worldwide. In this scoping review, we aim to investigate the Brazilian context for hemophilia regarding three predefined concepts: (i) clinical-epidemiological profile, (ii) burden of disease and (iii) patient journey and unmet needs. METHODS: Three questions in each concept guided the screening of references retrieved by systematic searches carried out in MEDLINE, LILACS and the Digital Library of Theses and Dissertations. Quantitative and qualitative studies conducted in Brazil from 2002 onwards were assessed for eligibility. MAIN RESULTS: Ninety-two studies were included. A total of 66 studies addressed the concept "Clinical-epidemiological profile", 31 investigated the concept of "Burden of disease" and 26 addressed the concept "Patient journey and unmet needs". Based on these studies, pain and arthropathy affect a substantial proportion of the PwH, with physical functioning, pain and school or work being the domains of quality of life with the greatest impact. About 43 % to 82.6 % of the PwH are unemployed. Rates of inhibitor development are highly variable across studies, especially in hemophilia A. Adherence to prophylactic treatment ranges from 25 % to 72 %. The annualized bleeding rate is estimated at 2.4 ± 4.1. The barriers to treatment identified include distance to reference centers, lack of coordination of specialized and emergency care and restricted access to rehabilitation. CONCLUSIONS: Hemophilia poses a considerable burden on the PwH. Despite the available modalities of treatment, there are remaining unmet needs that should be addressed by researchers and policy makers in the future.

Inequalities in the access to diagnosis and treatment of obstructive sleep apnea in Brazil: a cross-sectional study.

STUDY OBJECTIVES: Obstructive sleep apnea (OSA) is highly prevalent, and positive airway pressure (PAP) therapy is the primary treatment. This study aimed to assess the diagnostic and PAP treatment resources for OSA within Brazil's Unified Health System and to identify potential inequalities and gaps. METHODS: A structured survey was sent to members of the Brazilian Sleep Association and the Brazilian Association of Sleep Medicine to identify sleep laboratories providing OSA diagnosis and/or treatment within Brazil's Unified Health System. The numbers of centers, care team structure, sleep studies availability, PAP accessibility, and follow-up services were characterized in all 5 Brazilian regions. RESULTS: Forty-seven centers were identified: Midwest (n = 4), Northeast (n = 10), North (n = 3), Southeast (n = 22), and South (n = 8). Most centers (70%) provided both OSA diagnosis and treatment, mainly in capitals and/or metropolises (87%). Ten out of 27 Brazilian Federal Units lacked sleep services for OSA management, with the North having the highest proportion of states without a sleep service (71%). The annual number of diagnostic exams for OSA was 14,932, with significant heterogeneity across regions (Midwest: 240; North: 400; Northeast: 3,564; South: 4,380; Southeast: 6,348). Mean waiting times for diagnosis and treatment were 11 and 8 months, respectively. Only 46% of PAP treatments were publicly funded, making legal injunctions and out-of-pocket expenditure common practices. CONCLUSIONS: This study revealed significant disparities in OSA diagnosis and treatment resources across Brazil, with the North region being particularly underserved. The findings underscore an urgent need for strategies to improve sleep care nationwide. CITATION: Drager LF, Santos RB, Pachito D, Albertini CS, Sert Kuniyoshi FH, Eckeli AL. Inequalities in the access to diagnosis and treatment of obstructive sleep apnea in Brazil: a cross-sectional study. J Clin Sleep Med. 2024;20(5):735-742.

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.

Adapting shift work schedules for sleep quality, sleep duration, and sleepiness in shift workers.

BACKGROUND: Shift work is associated with insufficient sleep, which can compromise worker alertness with ultimate effects on occupational health and safety. Adapting shift work schedules may reduce adverse occupational outcomes. OBJECTIVES: To assess the effects of shift schedule adaptation on sleep quality, sleep duration, and sleepiness among shift workers. SEARCH METHODS: We searched CENTRAL, PubMed, Embase, and eight other databases on 13 December 2020, and again on 20 April 2022, applying no language restrictions. SELECTION CRITERIA: We included randomised controlled trials (RCTs) and non-RCTs, including controlled before-after (CBA) trials, interrupted time series, and cross-over trials. Eligible trials evaluated any of the following shift schedule components. • Permanency of shifts • Regularity of shift changes • Direction of shift rotation • Speed of rotation • Shift duration • Timing of start of shifts • Distribution of shift schedule • Time off between shifts • Split shifts • Protected sleep • Worker participation We included studies that assessed sleep quality off-shift, sleep duration off-shift, or sleepiness during shifts. DATA COLLECTION AND ANALYSIS: Two review authors independently screened the titles and abstracts of the records recovered by the search, read through the full-text articles of potentially eligible studies, and extracted data. We assessed the risk of bias of included studies using the Cochrane risk of bias tool, with specific additional domains for non-randomised and cluster-randomised studies. For all stages, we resolved any disagreements by consulting a third review author. We presented the results by study design and combined clinically homogeneous studies in meta-analyses using random-effects models. We assessed the certainty of the evidence with GRADE. MAIN RESULTS: We included 11 studies with a total of 2125 participants. One study was conducted in a laboratory setting and was not considered for drawing conclusions on intervention effects. The included studies investigated different and often multiple changes to shift schedule, and were heterogeneous with respect to outcome measurement. Forward versus backward rotation Three CBA trials (561 participants) investigated the effects of forward rotation versus backward rotation. Only one CBA trial provided sufficient data for the quantitative analysis; it provided very low-certainty evidence that forward rotation compared with backward rotation did not affect sleep quality measured with the Basic Nordic Sleep Questionnaire (BNSQ; mean difference (MD) -0.20 points, 95% confidence interval (CI) -2.28 to 1.89; 62 participants) or sleep duration off-shift (MD -0.21 hours, 95% CI -3.29 to 2.88; 62 participants). However, there was also very low-certainty evidence that forward rotation reduced sleepiness during shifts measured with the BNSQ (MD -1.24 points, 95% CI -2.24 to -0.24; 62 participants). Faster versus slower rotation Two CBA trials and one non-randomised cross-over trial (341 participants) evaluated faster versus slower shift rotation. We were able to meta-analyse data from two studies. There was low-certainty evidence of no difference in sleep quality off-shift (standardised mean difference (SMD) -0.01, 95% CI -0.26 to 0.23) and very low-certainty evidence that faster shift rotation reduced sleep duration off-shift (SMD -0.26, 95% CI -0.51 to -0.01; 2 studies, 282 participants). The SMD for sleep duration translated to an MD of 0.38 hours' less sleep per day (95% CI -0.74 to -0.01). One study provided very low-certainty evidence that faster rotations decreased sleepiness during shifts measured with the BNSQ (MD -1.24 points, 95% CI -2.24 to -0.24; 62 participants). Limited shift duration (16 hours) versus unlimited shift duration Two RCTs (760 participants) evaluated 80-hour workweeks with maximum daily shift duration of 16 hours versus workweeks without any daily shift duration limits. There was low-certainty evidence that the 16-hour limit increased sleep duration off-shift (SMD 0.50, 95% CI 0.21 to 0.78; which translated to an MD of 0.73 hours' more sleep per day, 95% CI 0.30 to 1.13; 2 RCTs, 760 participants) and moderate-certainty evidence that the 16-hour limit reduced sleepiness during shifts, measured with the Karolinska Sleepiness Scale (SMD -0.29, 95% CI -0.44 to -0.14; which translated to an MD of 0.37 fewer points, 95% CI -0.55 to -0.17; 2 RCTs, 716 participants). Shorter versus longer shifts One RCT, one CBA trial, and one non-randomised cross-over trial (692 participants) evaluated shorter shift duration (eight to 10 hours) versus longer shift duration (two to three hours longer). There was very low-certainty evidence of no difference in sleep quality (SMD -0.23, 95% CI -0.61 to 0.15; which translated to an MD of 0.13 points lower on a scale of 1 to 5; 2 studies, 111 participants) or sleep duration off-shift (SMD 0.18, 95% CI -0.17 to 0.54; which translated to an MD of 0.26 hours' less sleep per day; 2 studies, 121 participants). The RCT and the non-randomised cross-over study found that shorter shifts reduced sleepiness during shifts, while the CBA study found no effect on sleepiness. More compressed versus more spread out shift schedules One RCT and one CBA trial (346 participants) evaluated more compressed versus more spread out shift schedules. The CBA trial provided very low-certainty evidence of no difference between the groups in sleep quality off-shift (MD 0.31 points, 95% CI -0.53 to 1.15) and sleep duration off-shift (MD 0.52 hours, 95% CI -0.52 to 1.56). AUTHORS' CONCLUSIONS: Forward and faster rotation may reduce sleepiness during shifts, and may make no difference to sleep quality, but the evidence is very uncertain. Very low-certainty evidence indicated that sleep duration off-shift decreases with faster rotation. Low-certainty evidence indicated that on-duty workweeks with shift duration limited to 16 hours increases sleep duration, with moderate-certainty evidence for minimal reductions in sleepiness. Changes in shift duration and compression of workweeks had no effect on sleep or sleepiness, but the evidence was of very low-certainty. No evidence is available for other shift schedule changes. There is a need for more high-quality studies (preferably RCTs) for all shift schedule interventions to draw conclusions on the effects of shift schedule adaptations on sleep and sleepiness in shift workers.

Insomnia symptoms during the covid-19 pandemic: a case-control study.

OBJECTIVE: To identify lifestyle-related, sociodemographic, and mental health characteristics of people with insomnia symptoms and people without insomnia during the pandemic. METHODS: A case-control study was conducted with data collected by snowball sampling using an online questionnaire. From November 2020 to April 2021, 6,360 people with a mean age of 43.5 years (SD = 14.3) participated in the survey. For this study, we considered 158 cases of insomnia disorder and 476 controls (three controls per case) randomly selected from the participants without sleep problems. RESULTS: The results of the comparative analysis between cases and controls showed that sleeping less than six hours daily (OR = 3.89; 95%CI 2.50-6.05), feeling sadness frequently (OR = 2.95; 95%CI 1.69-5.17), residing in metropolitan areas (OR = 1.71; 95%CI 1.04-2.84), being 40 years or older (OR = 1.93; 95%CI 1.22-3.06), and the interaction between occupation and poorer education (OR = 2.12; 95%CI 1.22-3.69) were predictors for symptoms of insomnia disorder during the pandemic. CONCLUSIONS: In addition to confirming the hypothesis that mental health problems are associated with insomnia symptoms, the results point to insomnia as an important outcome for studies on the effects of unemployment, vulnerability and low education of the population, especially in large cities, highlighting that the effects of the crisis on health and the economy are extremely unequally distributed.

Pharmacological treatment for central sleep apnoea in adults.

BACKGROUND: The term central sleep apnoea (CSA) encompasses diverse clinical situations where a dysfunctional drive to breathe leads to recurrent respiratory events, namely apnoea (complete absence of ventilation) and hypopnoea sleep (insufficient ventilation) during sleep. Studies have demonstrated that CSA responds to some extent to pharmacological agents with distinct mechanisms, such as sleep stabilisation and respiratory stimulation. Some therapies for CSA are associated with improved quality of life, although the evidence on this association is uncertain. Moreover, treatment of CSA with non-invasive positive pressure ventilation is not always effective or safe and may result in a residual apnoea-hypopnoea index. OBJECTIVES: To evaluate the benefits and harms of pharmacological treatment compared with active or inactive controls for central sleep apnoea in adults. SEARCH METHODS: We used standard, extensive Cochrane search methods. The latest search date was 30 August 2022. SELECTION CRITERIA: We included parallel and cross-over randomised controlled trials (RCTs) that evaluated any type of pharmacological agent compared with active controls (e.g. other medications) or passive controls (e.g. placebo, no treatment or usual care) in adults with CSA as defined by the International Classification of Sleep Disorders 3rd Edition. We did not exclude studies based on the duration of intervention or follow-up. We excluded studies focusing on CSA due to periodic breathing at high altitudes. DATA COLLECTION AND ANALYSIS: We used standard Cochrane methods. Our primary outcomes were central apnoea-hypopnoea index (cAHI), cardiovascular mortality and serious adverse events. Our secondary outcomes were quality of sleep, quality of life, daytime sleepiness, AHI, all-cause mortality, time to life-saving cardiovascular intervention, and non-serious adverse events. We used GRADE to assess certainty of evidence for each outcome. MAIN RESULTS: We included four cross-over RCTs and one parallel RCT, involving a total of 68 participants. Mean age ranged from 66 to 71.3 years and most participants were men. Four trials recruited people with CSA associated with heart failure, and one study included people with primary CSA. Types of pharmacological agents were acetazolamide (carbonic anhydrase inhibitor), buspirone (anxiolytic), theophylline (methylxanthine derivative) and triazolam (hypnotic), which were given for between three days and one week. Only the study on buspirone reported a formal evaluation of adverse events. These events were rare and mild. No studies reported serious adverse events, quality of sleep, quality of life, all-cause mortality, or time to life-saving cardiovascular intervention. Carbonic anhydrase inhibitors versus inactive control Results were from two studies of acetazolamide versus placebo (n = 12) and acetazolamide versus no acetazolamide (n = 18) for CSA associated with heart failure. One study reported short-term outcomes and the other reported intermediate-term outcomes. We are uncertain whether carbonic anhydrase inhibitors compared to inactive control reduce cAHI in the short term (mean difference (MD) -26.00 events per hour, 95% CI -43.84 to -8.16; 1 study, 12 participants; very low certainty). Similarly, we are uncertain whether carbonic anhydrase inhibitors compared to inactive control reduce AHI in the short term (MD -23.00 events per hour, 95% CI -37.70 to 8.30; 1 study, 12 participants; very low certainty) or in the intermediate term (MD -6.98 events per hour, 95% CI -10.66 to -3.30; 1 study, 18 participants; very low certainty). The effect of carbonic anhydrase inhibitors on cardiovascular mortality in the intermediate term was also uncertain (odds ratio (OR) 0.21, 95% CI 0.02 to 2.48; 1 study, 18 participants; very low certainty). Anxiolytics versus inactive control Results were based on one study of buspirone versus placebo for CSA associated with heart failure (n = 16). The median difference between groups for cAHI was -5.00 events per hour (IQR -8.00 to -0.50), the median difference for AHI was -6.00 events per hour (IQR -8.80 to -1.80), and the median difference on the Epworth Sleepiness Scale for daytime sleepiness was 0 points (IQR -1.0 to 0.00). Methylxanthine derivatives versus inactive control Results were based on one study of theophylline versus placebo for CSA associated with heart failure (n = 15). We are uncertain whether methylxanthine derivatives compared to inactive control reduce cAHI (MD -20.00 events per hour, 95% CI -32.15 to -7.85; 15 participants; very low certainty) or AHI (MD -19.00 events per hour, 95% CI -30.27 to -7.73; 15 participants; very low certainty). Hypnotics versus inactive control Results were based on one trial of triazolam versus placebo for primary CSA (n = 5). Due to very serious methodological limitations and insufficient reporting of outcome measures, we were unable to draw any conclusions regarding the effects of this intervention. AUTHORS' CONCLUSIONS: There is insufficient evidence to support the use of pharmacological therapy in the treatment of CSA. Although small studies have reported positive effects of certain agents for CSA associated with heart failure in reducing the number of respiratory events during sleep, we were unable to assess whether this reduction may impact the quality of life of people with CSA, owing to scarce reporting of important clinical outcomes such as sleep quality or subjective impression of daytime sleepiness. Furthermore, the trials mostly had short-term follow-up. There is a need for high-quality trials that evaluate longer-term effects of pharmacological interventions.

Legal action for access to resources inefficiently made available in health care systems in Brazil: a case study on obstructive sleep apnea.

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.

Insomnia symptoms during the covid-19 pandemic: a case-control study / Sintomas de insônia durante a pandemia de covid-19: um estudo caso-controle

ABSTRACT OBJECTIVE To identify lifestyle-related, sociodemographic, and mental health characteristics of people with insomnia symptoms and people without insomnia during the pandemic. METHODS A case-control study was conducted with data collected by snowball sampling using an online questionnaire. From November 2020 to April 2021, 6,360 people with a mean age of 43.5 years (SD = 14.3) participated in the survey. For this study, we considered 158 cases of insomnia disorder and 476 controls (three controls per case) randomly selected from the participants without sleep problems. RESULTS The results of the comparative analysis between cases and controls showed that sleeping less than six hours daily (OR = 3.89; 95%CI 2.50-6.05), feeling sadness frequently (OR = 2.95; 95%CI 1.69-5.17), residing in metropolitan areas (OR = 1.71; 95%CI 1.04-2.84), being 40 years or older (OR = 1.93; 95%CI 1.22-3.06), and the interaction between occupation and poorer education (OR = 2.12; 95%CI 1.22-3.69) were predictors for symptoms of insomnia disorder during the pandemic. CONCLUSIONS In addition to confirming the hypothesis that mental health problems are associated with insomnia symptoms, the results point to insomnia as an important outcome for studies on the effects of unemployment, vulnerability and low education of the population, especially in large cities, highlighting that the effects of the crisis on health and the economy are extremely unequally distributed.

RESUMO OBJETIVO Identificar características relacionadas ao estilo de vida, sociodemográficas e saúde mental de pessoas com sintomas de insônia e pessoas sem insônia durante a pandemia. MÉTODOS A partir de dados coletados por amostragem em bola de neve, por meio de um questionário online foi realizado um estudo caso-controle. Durante o período de novembro de 2020 a abril de 2021, 6.360 pessoas com idade média de 43,5 anos (DP = 14,3) participaram da pesquisa. No presente estudo, foram considerados 158 casos de transtorno de insônia e 476 controles (três controles por caso) selecionados aleatoriamente dentre os participantes sem problemas de sono. RESULTADOS Os resultados da análise comparativa entre casos e controles mostraram que dormir menos de seis horas diárias (OR = 3,89; IC95% 2,50-6,05), sentir tristeza frequentemente (OR = 2,95; IC95% 1,69-5,17), residir em metrópoles (OR = 1,71; IC95% 1,04-2,84), estar com 40 anos ou mais (OR = 1,93; IC95% 1,22-3,06) e a interação entre ocupação e escolaridade mais precária (OR = 2,12; IC95% 1,22-3,69) foram fatores preditores para sintomas de transtorno de insônia durante a pandemia. CONCLUSÕES Além da confirmação da hipótese de que problemas de saúde mental estão associados a sintomas de insônia, os resultados apontam para a insônia como um desfecho importante para estudos sobre efeitos do desemprego, vulnerabilidade e baixa escolaridade da população, sobretudo nas grandes metrópoles, ressaltando que os efeitos da crise sobre a saúde e a economia são distribuídos de forma extremamente desiguais.

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.

Teleconsultations in neurology in a universal health system amid COVID-19: a descriptive study.

OBJECTIVE: This study aimed to characterize teleconsultations in neurology executed by Regula+Brasil project in Recife, a capital city in northeastern Brazil. METHODS: A descriptive study carried out by four private hospitals, in a partnership with the Ministry of Health in Brazil. Teleconsultation was performed preferably in the video modality. Conditions eligible for teleconsultation were headache, epilepsy, and cerebrovascular disorders. Period of analysis was May to September 2020. RESULTS: A total of 243 teleconsultations were analyzed, of which 76.95% was a first appointment. In 48.97% of cases, the teleconsultation represented the first opportunity for the patient to be consulted with the specialist. Among cases of first appointment, 20.16% were further referred to a face-to-face consultation and 21.81% could be redirected to primary health care. Headache disorders were the most predominant clinical conditions. CONCLUSIONS: The implementation and development of telemedicine by Regula+Brasil during the COVID-19 pandemic represented an opportunity to assess the value of having teleconsultations added along the line of care from primary care to a medical specialty, promoting the coordination of care across different levels of complexity of care in the health system and improving access to specialized care.

Non-invasive positive pressure ventilation for central sleep apnoea in adults.

BACKGROUND: Central sleep apnoea (CSA) is characterised by abnormal patterns of ventilation during sleep due to a dysfunctional drive to breathe. Consequently, people with CSA may present poor sleep quality, sleep fragmentation, inattention, fatigue, daytime sleepiness, and reduced quality of life. OBJECTIVES: To assess the effectiveness and safety of non-invasive positive pressure ventilation (NIPV) for the treatment of adults with CSA. SEARCH METHODS: We searched the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, Embase, and Scopus on 6 September 2021. We applied no restrictions on language of publication. We also searched clinical trials registries for ongoing and unpublished studies, and scanned the reference lists of included studies to identify additional studies. SELECTION CRITERIA: We included randomised controlled trials (RCTs) reported in full text, those published as abstract only, and unpublished data. DATA COLLECTION AND ANALYSIS: Two review authors independently selected studies for inclusion, extracted data, and assessed risk of bias of the included studies using the Cochrane risk of bias tool version 1.0, and the certainty of the evidence using the GRADE approach. In the case of disagreement, a third review author was consulted. MAIN RESULTS: We included 15 RCTs with a total of 1936 participants, ranging from 10 to 1325 participants. All studies had important methodological limitations. We assessed most studies (11 studies) as at high risk of bias for at least one domain, and all studies as at unclear risk of bias for at least two domains. The trials included participants aged > 18 years old, of which 70% to 100% were men, who were followed from one week to 60 months. The included studies assessed the effects of different modes of NIPV and CSA. Most participants had CSA associated with chronic heart failure. Because CSA encompasses a variety of causes and underlying clinical conditions, data were carefully analysed, and different conditions and populations were not pooled. The findings for the primary outcomes for the seven evaluated comparisons are presented below.  Continuous positive airway pressure (CPAP) plus best supportive care versus best supportive care in CSA associated with chronic heart failure In the short term, CPAP plus best supportive care may reduce central apnoea hypopnoea index (AHI) (mean difference (MD) -14.60, 95% confidence interval (CI) -20.11 to -9.09; 1 study; 205 participants). However, CPAP plus best supportive care may result in little to no difference in cardiovascular mortality compared to best supportive care alone. The evidence for the effect of CPAP plus best supportive care on all-cause mortality is very uncertain. No adverse effects were observed with CPAP, and the results for adverse events in the best supportive care group were not reported. Adaptive servo ventilation (ASV) versus CPAP in CSA associated with chronic heart failure The evidence is very uncertain about the effect of ASV versus CPAP on quality of life evaluated in both the short and medium term. Data on adverse events were not reported, and it is not clear whether data were sought but not found. ASV versus bilevel ventilation in CSA associated with chronic heart failure In the short term, ASV may result in little to no difference in central AHI. No adverse events were detected with ASV, and the results for adverse events in the bilevel ventilation group were not reported. ASV plus best supportive care versus best supportive care in CSA associated with chronic heart failure In the medium term, ASV plus best supportive care may reduce AHI compared to best supportive care alone (MD -20.30, 95% CI -28.75 to -11.85; 1 study; 30 participants). In the long term, ASV plus best supportive care likely increases cardiovascular mortality compared to best supportive care (risk ratio (RR) 1.25, 95% CI 1.04, 1.49; 1 study; 1325 participants). The evidence suggests that ASV plus best supportive care may result in little to no difference in quality of life in the short, medium, and long term, and in all-cause mortality in the medium and long term. Data on adverse events were evaluated but not reported. ASV plus best supportive care versus best supportive care in CSA with acute heart failure with preserved ejection fraction Only adverse events were reported for this comparison, and no adverse events were recorded in either group. ASV versus CPAP maintenance in CPAP-induced CSA In the short term, ASV may slightly reduce central AHI (MD -4.10, 95% CI -6.67 to -1.53; 1 study; 60 participants), but may result in little to no difference in quality of life. Data on adverse events were not reported, and it is not clear whether data were sought but not found. ASV versus bilevel ventilation in CPAP-induced CSA In the short term, ASV may slightly reduce central AHI (MD -8.70, 95% CI -11.42 to -5.98; 1 study; 30 participants) compared to bilevel ventilation. Data on adverse events were not reported, and it is not clear whether data were sought but not found. AUTHORS' CONCLUSIONS: CPAP plus best supportive care may reduce central AHI in people with CSA associated with chronic heart failure compared to best supportive care alone. Although ASV plus best supportive care may reduce AHI in people with CSA associated with chronic heart failure, it likely increases cardiovascular mortality in these individuals. In people with CPAP-induced CSA, ASV may slightly reduce central AHI compared to bilevel ventilation and to CPAP. In the absence of data showing a favourable impact on meaningful patient-centred outcomes and defining clinically important differences in outcomes in CSA patients, these findings need to be interpreted with caution. Considering the level of certainty of the available evidence and the heterogeneity of participants with CSA, we could draw no definitive conclusions, and further high-quality trials focusing on patient-centred outcomes, such as quality of life, quality of sleep, and longer-term survival, are needed to determine whether one mode of NIPV is better than another or than best supportive care for any particular CSA patient group.

Teleconsultations in neurology in a universal health system amid COVID-19: a descriptive study

SUMMARY OBJECTIVE: This study aimed to characterize teleconsultations in neurology executed by Regula+Brasil project in Recife, a capital city in northeastern Brazil. METHODS: A descriptive study carried out by four private hospitals, in a partnership with the Ministry of Health in Brazil. Teleconsultation was performed preferably in the video modality. Conditions eligible for teleconsultation were headache, epilepsy, and cerebrovascular disorders. Period of analysis was May to September 2020. RESULTS: A total of 243 teleconsultations were analyzed, of which 76.95% was a first appointment. In 48.97% of cases, the teleconsultation represented the first opportunity for the patient to be consulted with the specialist. Among cases of first appointment, 20.16% were further referred to a face-to-face consultation and 21.81% could be redirected to primary health care. Headache disorders were the most predominant clinical conditions. CONCLUSIONS: The implementation and development of telemedicine by Regula+Brasil during the COVID-19 pandemic represented an opportunity to assess the value of having teleconsultations added along the line of care from primary care to a medical specialty, promoting the coordination of care across different levels of complexity of care in the health system and improving access to specialized care.

COVID-19 pandemic is associated with increased sleep disturbances and mental health symptoms but not help-seeking: a cross-sectional nation-wide study.

Objective: This study aimed firstly to describe sleep-related and mental health symptoms before and during the COVID-19 pandemic in a national-wide sample and, secondly, to verify attitudes towards help-seeking to treat these symptoms. Material and Methods: Data were collected through an online questionnaire sent through the Brazilian Sleep Association's social media. The questionnaire included sociodemographic and sleep aspects questions currently and before the pandemic period. In addition, the survey addressed current and previous anxiety, depression, and burnout symptoms. The outcome help-seeking was addressed in the questionnaire as well by a single question asked when the participant reported mental or sleep problems. Results: The study covered 6,360 participants, mean age 43.5 years (SD=14.3), 76.7% female and 63.7% with undergraduate or higher degree filled out the survey. Seventy percent of participants reported sleep disturbances and 80% reported symptoms of anxiety during the pandemic. Help-seeking behavior was found only in one third of them. Hours of sleep reduced from 7.12 to 6.2h, which can be related with the increase in 28.2% of dissatisfaction with sleep duration during the pandemic. The highest frequency of complaints related to sleep was difficulty to fall asleep three or more times a week (going from 27.6% before the pandemic to 58.9% during the pandemic; p<0.001). Moreover, it was observed that help-seeking was more prevalent in men than women, and more in younger participants than in older ones. Conclusion: There was an increase of sleep and mental self-reported problems during the pandemic, which was not followed by help-seeking.

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.

Micro-Costing of a Remotely Operated Referral Management System to Secondary Care in the Unified Health System in Brazil.

Referral of cases from primary to secondary care in the Brazilian public healthcare system is one of the most important issues to be tackled. Telehealth strategies have been shown effective in avoiding unnecessary referrals. The objective of this study was to estimate cost per referred case by a remotely operated referral management system to further inform the decision making on the topic. Analysis of cost by applying time-driven activity-based costing. Cost analyses included comparisons between medical specialties, localities for which referrals were being conducted, and periods of time. Cost per referred case across localities ranged from R$ 5.70 to R$ 8.29. Cost per referred case across medical specialties ranged from R$ 1.85 to R$ 8.56. Strategies to optimize the management of referral cases to specialized care in public healthcare systems are still needed. Telehealth strategies may be advantageous, with cost estimates across localities ranging from R$ 5.70 to R$ 8.29, with additional observed variability related to the type of medical specialty.

Micro-Costing of a Remotely Operated Referral Management System to Secondary Care in the Unified Health System in Brazil / Microcusteio de um Sistema de Regulação Remoto para o Cuidado Especializado no Sistema Único de Saúde no Brasil

Abstract Referral of cases from primary to secondary care in the Brazilian public healthcare system is one of the most important issues to be tackled. Telehealth strategies have been shown effective in avoiding unnecessary referrals. The objective of this study was to estimate cost per referred case by a remotely operated referral management system to further inform the decision making on the topic. Analysis of cost by applying time-driven activity-based costing. Cost analyses included comparisons between medical specialties, localities for which referrals were being conducted, and periods of time. Cost per referred case across localities ranged from R$ 5.70 to R$ 8.29. Cost per referred case across medical specialties ranged from R$ 1.85 to R$ 8.56. Strategies to optimize the management of referral cases to specialized care in public healthcare systems are still needed. Telehealth strategies may be advantageous, with cost estimates across localities ranging from R$ 5.70 to R$ 8.29, with additional observed variability related to the type of medical specialty.

Resumo O encaminhamento de casos da atenção primária para a secundária no Sistema Único Brasileiro é uma das questões mais importantes a ser enfrentada. As estratégias de telessaúde têm se mostrado eficazes para evitar encaminhamentos desnecessários. O objetivo deste estudo foi estimar o custo por caso encaminhado por meio de um sistema de gerenciamento de referenciamentos operado remotamente para subsidiar a tomada de decisão sobre o tema. Análise de custo por meio da aplicação de custeio baseado em atividades orientado pelo tempo (time-driven activity-based costing ou TDABC). As análises de custo incluíram comparações entre especialidades médicas, localidades para as quais os encaminhamentos estavam sendo conduzidos e períodos de tempo. O custo por referenciamento em todas as localidades variou entre R$ 5,70 a R$ 8,29. O custo por referenciamento nas especialidades médicas variou entre R$ 1,85 a R$ 8,56. Estratégias para otimizar a gestão dos referenciamentos para a atenção especializada nos sistemas públicos de saúde ainda são necessárias. As estratégias de telessaúde podem ser vantajosas, com estimativas de custo entre as localidades variando entre R$ 5,70 a R$ 8,29, com variabilidade adicional observada relacionada ao tipo de especialidade médica.

Assessing the quality of evidence in studies estimating prevalence of exposure to occupational risk factors: The QoE-SPEO approach applied in the systematic reviews 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) have produced the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury (WHO/ILO Joint Estimates). For these, systematic reviews of studies estimating the prevalence of exposure to selected occupational risk factors have been conducted to provide input data for estimations of the number of exposed workers. A critical part of systematic review methodology is to assess the quality of evidence across studies. In this article, we present the approach applied in these WHO/ILO systematic reviews for performing such assessments on studies of prevalence of exposure. It is called the Quality of Evidence in Studies estimating Prevalence of Exposure to Occupational risk factors (QoE-SPEO) approach. We describe QoE-SPEO's development to date, demonstrate its feasibility reporting results from pilot testing and case studies, note its strengths and limitations, and suggest how QoE-SPEO should be tested and developed further. METHODS: Following a comprehensive literature review, and using expert opinion, selected existing quality of evidence assessment approaches used in environmental and occupational health were reviewed and analysed for their relevance to prevalence studies. Relevant steps and components from the existing approaches were adopted or adapted for QoE-SPEO. New steps and components were developed. We elicited feedback from other systematic review methodologists and exposure scientists and reached consensus on the QoE-SPEO approach. Ten individual experts pilot-tested QoE-SPEO. To assess inter-rater agreement, we counted ratings of expected (actual and non-spurious) heterogeneity and quality of evidence and calculated a raw measure of agreement (Pi) between individual raters and rater teams for the downgrade domains. Pi ranged between 0.00 (no two pilot testers selected the same rating) and 1.00 (all pilot testers selected the same rating). Case studies were conducted of experiences of QoE-SPEO's use in two WHO/ILO systematic reviews. RESULTS: We found no existing quality of evidence assessment approach for occupational exposure prevalence studies. We identified three relevant, existing approaches for environmental and occupational health studies of the effect of exposures. Assessments using QoE-SPEO comprise three steps: (1) judge the level of expected heterogeneity (defined as non-spurious variability that can be expected in exposure prevalence, within or between individual persons, because exposure may change over space and/or time), (2) assess downgrade domains, and (3) reach a final rating on the quality of evidence. Assessments are conducted using the same five downgrade domains as the Grading of Recommendations Assessment, Development and Evaluation (GRADE) approach: (a) risk of bias, (b) indirectness, (c) inconsistency, (d) imprecision, and (e) publication bias. For downgrade domains (c) and (d), the assessment varies depending on the level of expected heterogeneity. There are no upgrade domains. The QoE-SPEO's ratings are "very low", "low", "moderate", and "high". To arrive at a final decision on the overall quality of evidence, the assessor starts at "high" quality of evidence and for each domain downgrades by one or two levels for serious concerns or very serious concerns, respectively. In pilot tests, there was reasonable agreement in ratings for expected heterogeneity; 70% of raters selected the same rating. Inter-rater agreement ranged considerably between downgrade domains, both for individual rater pairs (range Pi: 0.36-1.00) and rater teams (0.20-1.00). Sparse data prevented rigorous assessment of inter-rater agreement in quality of evidence ratings. CONCLUSIONS: We present QoE-SPEO as an approach for assessing quality of evidence in prevalence studies of exposure to occupational risk factors. It has been developed to its current version (as presented here), has undergone pilot testing, and was applied in the systematic reviews for the WHO/ILO Joint Estimates. While the approach requires further testing and development, it makes steps towards filling an identified gap, and progress made so far can be used to inform future work in this area.

Assessor burden, inter-rater agreement and user experience of the RoB-SPEO tool for assessing risk of bias in studies estimating prevalence of exposure to occupational risk factors: An analysis from the WHO/ILO Joint Estimates of the Work-related Burden of Disease and Injury.

BACKGROUND: As part of the development of the World Health Organization (WHO)/International Labour Organization (ILO) Joint Estimates of the Work-related Burden of Disease and Injury, WHO and ILO carried out several systematic reviews to determine the prevalence of exposure to selected occupational risk factors. Risk of bias assessment for individual studies is a critical step of a systematic review. No tool existed for assessing the risk of bias in prevalence studies of exposure to occupational risk factors, so WHO and ILO developed and pilot tested the RoB-SPEO tool for this purpose. Here, we investigate the assessor burden, inter-rater agreement, and user experience of this new instrument, based on the abovementioned WHO/ILO systematic reviews. METHODS: Twenty-seven individual experts applied RoB-SPEO to assess risk of bias. Four systematic reviews provided a total of 283 individual assessments, carried out for 137 studies. For each study, two or more assessors independently assessed risk of bias across the eight RoB-SPEO domains selecting one of RoB-SPEO's six ratings (i.e., "low", "probably low", "probably high", "high", "unclear" or "cannot be determined"). Assessors were asked to report time taken (i.e. indicator of assessor burden) to complete each assessment and describe their user experience. To gauge assessor burden, we calculated the median and inter-quartile range of times taken per individual risk of bias assessment. To assess inter-rater reliability, we calculated a raw measure of inter-rater agreement (Pi) for each RoB-SPEO domain, between Pi = 0.00, indicating no agreement and Pi = 1.00, indicating perfect agreement. As subgroup analyses, Pi was also disaggregated by systematic review, assessor experience with RoB-SPEO (≤10 assessments versus > 10 assessments), and assessment time (tertiles: ≤25 min versus 26-66 min versus ≥ 67 min). To describe user experience, we synthesised the assessors' comments and recommendations. RESULTS: Assessors reported a median of 40 min to complete one assessment (interquartile range 21-120 min). For all domains, raw inter-rater agreement ranged from 0.54 to 0.82. Agreement varied by systematic review and assessor experience with RoB-SPEO between domains, and increased with increasing assessment time. A small number of users recommended further development of instructions for selected RoB-SPEO domains, especially bias in selection of participants into the study (domain 1) and bias due to differences in numerator and denominator (domain 7). DISCUSSION: Overall, our results indicated good agreement across the eight domains of the RoB-SPEO tool. The median assessment time was comparable to that of other risk of bias tools, indicating comparable assessor burden. However, there was considerable variation in time taken to complete assessments. Additional time spent on assessments may improve inter-rater agreement. Further development of the RoB-SPEO tool could focus on refining instructions for selected RoB-SPEO domains and additional testing to assess agreement for different topic areas and with a wider range of assessors from different research backgrounds.

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.

Insomnia episodes, new-onset pharmacological treatments, and other sleep disturbances during the COVID-19 pandemic: a nationwide cross-sectional study in Brazilian health care professionals.

STUDY OBJECTIVES: To evaluate the impact of the coronavirus disease 2019 (COVID-19) pandemic on insomnia and other sleep disturbances in health care professionals. METHODS: A survey was distributed using social media and organizational emails to Brazilian active health care professionals during the COVID-19 outbreak. We explored potential associated factors including age, sex, occupation, workplace, work hours, income, previous infection with COVID-19, recent/current contact with COVID-19 patients, regional number of incident deaths, anxiety, and burnout. We evaluated new-onset/previous insomnia worsening episodes (primary outcome), new pharmacological treatments, sleep quality, duration, nightmares, and snoring (secondary outcomes). RESULTS: A total of 4,384 health professionals from all regions of the country were included in the analysis (44 ± 12 years, 76% females, 53.8% physicians). Overall, 55.7% were assisting patients with COVID-19, and 9.2% had a previous COVID-19 infection. The primary outcome occurred in 32.9% of respondents in parallel to 13% new pharmacological treatments for insomnia. The sleep quality worsened for 61.4%, while 43.5% and 22.8% reported ≥ 1-hour sleep duration reduction and worsening or new-onset nightmares, respectively. Multivariate analyses showed that age (odds ratio [OR]: 1.008; 95% confidence interval [CI] 1.001-1.015), females (OR: 1.590; 95% CI 1.335-1.900), weight change (decrease: OR: 1.772; 95% CI 1.453-2.161; increase: OR: 1.468; 95% CI 1.249-1.728), prevalent anxiety (OR: 3.414; 95% CI 2.954-3.948), new-onset burnout (OR: 1.761; 95% CI 1.489-2.083), family income reduction > 30% (OR: 1.288; 95% CI 1.069-1.553), and assisting patients with COVID-19 (OR: 1.275; 95% CI 1.081-1.506) were independently associated with new-onset or worsening of previous insomnia episodes. CONCLUSIONS: We observed a huge burden of insomnia episodes and other sleep disturbances in health care professionals during the COVID-19 pandemic. CITATION: Drager LF, Pachito DV, Moreno CRC, et al. Insomnia episodes, new-onset pharmacological treatments, and other sleep disturbances during the COVID-19 pandemic: a nationwide cross-sectional study in Brazilian health care professionals. J Clin Sleep Med. 2022;18(2):373-382.