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BACKGROUND AND OBJECTIVES: Case 2 best-worst scaling (BWS-2) is an increasingly popular method to elicit patient preferences. Because BWS-2 potentially has a lower cognitive burden compared with discrete choice experiments, the aim of this study was to compare treatment preference weights and relative importance scores.METHODS: Patients with neuromuscular diseases completed an online survey at two different moments in time, completing one method per occasion. Patients were randomly assigned to either first a discrete choice experiment or BWS-2. Attributes included: muscle strength, energy endurance, balance, cognition, chance of blurry vision, and chance of liver damage. Multinomial logit was used to calculate overall relative importance scores and latent class logit was used to estimate heterogeneous preference weights and to calculate the relative importance scores of the attributes for each latent class.RESULTS: A total of 140 patients were included for analyses. Overall relative importance scores showed differences in attribute importance rankings between a discrete choice experiment and BWS-2. Latent class analyses indicated three latent classes for both methods, with a specific class in both the discrete choice experiment and BWS-2 in which (avoiding) liver damage was the most important attribute. Ex-post analyses showed that classes differed in sex, age, level of education, and disease status. The discrete choice experiment was easier to understand compared with BWS-2.CONCLUSIONS: This study showed that using a discrete choice experiment and BWS-2 leads to different outcomes, both in preference weights as well as in relative importance scores, which might have been caused by the different framing of risks in BWS-2. However, a latent class analysis revealed similar latent classes between methods. Careful consideration about method selection is required, while keeping the specific decision context in mind and pilot testing the methods.

ObjectivesThe COVID-19 pandemic forms an unprecedented public health, economic, and social crisis. Uptake of vaccination is critical for controlling the pandemic. Nevertheless, vaccination hesitancy is considerable, requiring policies to promote uptake. We investigate Dutch citizens’ preferences for policies that aim to promote vaccination through facilitating choice of vaccination, profiling it as the norm, making vaccination more attractive through rewards, or punishing people who reject vaccination.MethodsWe conducted a discrete choice experiment in which 747 respondents were asked to choose between policies to promote vaccination uptake and their impacts on the number of deaths, people with permanent health problems, households with income loss, and a tax increase.ResultsRespondents generally had a negative preference for policies that promote vaccination. They particularly disliked policies that punish those who reject the vaccine and were more favorable toward policies that reward vaccination, such as awarding additional rights to vaccinated individuals through vaccination passports. Respondents who reject vaccination were in general much more negative about the policy options than respondents who consider accepting the vaccine. Nevertheless, vaccination passports are supported by both respondents who accept the vaccine, those who reject vaccination, and those who are unsure about vaccination.ConclusionsThis study provides concrete directions for governments attempting to increase the vaccination uptake in ways that are supported by the public. Our results could encourage policy makers to focus on policy options that make vaccination easier and reward people who take the vaccine, as especially the implementation of vaccination passports was supported.

BackgroundDuring the COVID-19 pandemic cancer patients might have experienced delays in screening, diagnosis and/or treatment. A systematic review was conducted to give an overview of the effects of COVID-19 induced delays in oncological care on the physical and mental health outcomes of cancer patients.MethodsMEDLINE and EMBASE databases were searched for articles on the effects of COVID-19 induced delays on physical and mental health outcomes.ResultsOut of 1333 papers, eighteen observational, and twelve modelling studies were included. In approximately half of the studies, tumor stage distribution differed during the pandemic compared to before the pandemic. Modelling studies predicted that the estimated increase in the number of deaths ranged from -0.04 to 30%, and the estimated reduction in survival ranged from 0.4 to 35%. Varying results on the impact on mental health, e.g. anxiety and depression, were seen.ConclusionsDue to large methodological discrepancies between the studies and the varying results, the effect of COVID-19 induced delays on the physical and mental health outcomes of cancer patients remains uncertain. While modelling studies estimated an increase in mortality, observational studies suggest that mortality might not increase to a large extent. More longitudinal observational data from the pandemic period is needed for more conclusive results.

Introduction: Ensuring patients have enough information about healthcare choices prior to completing a preference study is necessary to support the validity of the findings. Patients are commonly informed using text-based information with supporting graphics. Video-based information may be more engaging for the general patient population. This study aimed to assess (1) the impact that educating patients using video-based educational materials with a voiceover has on patient preferences compared to traditional text, and (2) whether this impact is consistent between two countries. Materials and Methods: A video-based educational tool was developed to inform patients prior to completing a discrete choice experiment assessing preferences for glucose monitors. Patients with diabetes from the Netherlands and Poland were recruited through an online research panel. Respondents were randomised to receive information in either a text or a video with animations and a voiceover. Data were analysed using a mixed-logit model. Results: N = 981 completed surveys were analysed from the Netherlands (n = 459) and Poland (n = 522). Differences were found between the countries, but no interpretable pattern of differences was found between the two types of educational materials. Patients spent less time in the educational material than would be necessary to fully review all of the content. Conclusions: Simply providing educational material in a video with animations and voiceovers does not necessarily lead to better engagement from respondents or different preference outcomes in a sample of diabetes patients when compared to text. Increasing engagement with educational materials should be a topic of future research for those conducting patient preference research as no amount of educational material will be helpful if respondents do not access it.

Background: The COVID-19 pandemic had a major impact on the continuity of healthcare provision. Appointments, treatments and surgeries for non-COVID patients were often delayed, with associated health losses for patients involved. Objective: To develop a method to quantify the health impact of delayed elective care for non-COVID patients. Methods: A model was developed that estimated the backlog of surgical procedures in 2020 and 2021 using hospital registry data. Quality-adjusted life years (QALYs) were obtained from the literature to estimate the non-generated QALYs related to the backlog. In sensitivity analyses QALY values were varied by type of patient prioritization. Scenario analyses for future increased surgical capacity were performed. Results: In 2020 and 2021 an estimated total of 305,374 elective surgeries were delayed. These delays corresponded with 319,483 non-generated QALYs. In sensitivity analyses where QALYs varied by type of patient prioritization, non-generated QALYs amounted to 150,973 and 488,195 QALYs respectively. In scenario analyses for future increased surgical capacity in 2022–2026, the non-generated QALYs decreased to 311,220 (2% future capacity increase per year) and 300,710 (5% future capacity increase per year). Large differences exist in the extent to which different treatments contributed to the total health losses. Conclusions: The method sheds light on the indirect harm related to the COVID-19 pandemic. The results can be used for policy evaluations of COVID-19 responses, in preparations for future waves or other pandemics and in prioritizing the allocation of resources for capacity increases.

Background Recent attempts of active disinvestment (i.e. withdrawal of reimbursement by means of a policy decision) of reimbursed healthcare interventions in the Netherlands have differed in their outcome: some attempts were successful, with interventions actually being disinvested. Other attempts were terminated at some point, implying unsuccessful disinvestment. This study aimed to obtain insight into recent active disinvestment processes, and to explore what aspects affect their outcome. Methods Semi-structured interviews were conducted from January to December 2018 with stakeholders (e.g. patients, policymakers, physicians) who were involved in the policy process of five cases for which the full or partial withdrawal of reimbursement was considered in the Netherlands between 2007 and 2017: benzodiazepines, medication for Fabry disease, quit smoking programme, psychoanalytic therapy and maternity care assistance. These cases covered both interventions that were eventually disinvested and interventions for which reimbursement was maintained after consideration. Interviews were transcribed verbatim, double coded and analyzed using thematic analysis. Results The 37 interviews showed that support for disinvestment from stakeholders, especially from healthcare providers and policymakers, strongly affected the outcome of the disinvestment process. Furthermore, the institutional role of stakeholders as legitimized by the Dutch health insurance system, their financial interests in maintaining or discontinuing reimbursement, and the possibility to relieve the consequences of disinvestment for current patients affected the outcome of the disinvestment process as well. A poor organization of patient groups may make it difficult for patients to exert pressure, which may contribute to successful disinvestment. No evidence was found of a consistent role of the formal Dutch package criteria (i.e. effectiveness, cost-effectiveness, necessity and feasibility) in active disinvestment processes. Conclusions Contextual factors as well as the possibility to relieve the consequences of disinvestment for current patients are important determinants of the outcome of active disinvestment processes. These results provide insight into active disinvestment processes and their determinants, and provide guidance to policymakers for a potentially more successful approach for future active disinvestment processes.

Background Acute otitis media (AOM) is among the most common paediatric conditions managed in primary care. Most recent estimates of the cost of AOM date from a decade ago and lack a full societal perspective. We therefore explored the societal cost of childhood AOM in the Netherlands within the setting of a trial comparing the effectiveness of an intervention aimed at educating general practitioners (GPs) about pain management in AOM compared to usual care. Methods Economic analysis alongside a cluster randomised controlled trial conducted between February 2015 and May 2018 in 37 practices (94 GPs). In total, 224 children with AOM were included of which 223 (99%) completed the trial (intervention: n = 94; control: n = 129). The cost of AOM due to health care costs, patient and family costs, and productivity losses by parent caregivers were retrieved from study diaries and primary care electronic health records, during 28-day follow-up. We calculated mean cost (€ and $) per AOM episode per patient with standard deviations (SD, in €) regardless of study group assignment because there was no clinical effect of the trial intervention. In sensitivity analysis, we calculated cost in the intervention and usual care group, after exclusion of extreme outliers. Results Mean total AOM cost per patient were €565.93 or $638.78 (SD €1071.01); nearly 90% of these costs were due to productivity losses experienced by parents. After exclusion of outliers, AOM cost was €526.70 or $594.50 (SD €987.96) and similar in the intervention and usual care groups: €516.10 or $582.53 (SD €949.69) and €534.55 or $603.36 (SD €920.55) respectively. Conclusions At €566 or $639 per episode, societal cost of AOM is higher than previously known and mainly driven by productivity losses by children’s parents. Considering its high incidence, AOM poses a significant economic burden that extends beyond direct medical costs. Trial registration Netherlands Trial Register no. NTR4920: http://www.trialregister.nl/trialreg/admin/rctview.asp?TC=4920.

Introduction It has become increasingly important to include patient preference information in decision-making processes for drug development. As neuromuscular disorders represent multisystem, debilitating, and progressive rare diseases with few treatment options, this study aimed to explore unmet health care needs and patient treatment preferences for two neuromuscular disorders, myotonic dystrophy type 1 (DM1) and mitochondrial myopathies (MM) to inform early stages of drug development. Methods Fifteen semi-structured interviews and five focus group discussions (FGDs) were held with DM1 and MM adult patients and caregivers. Topics discussed included (1) reasons for study participation; (2) disease signs/symptoms and their impact on daily lives; (3) top desired benefits; and (4) acceptability of risks and tolerance levels for a hypothetical new treatment. Data were analyzed following a thematic ‘code’ approach. Results A total of 52 participants representing a wide range of disease severities participated. ‘Muscle strength’ and ‘energy and endurance’ were the disease-related unmet needs most often mentioned. Additionally, improved ‘balance’, ‘cognition’ and ‘gut function’ were the top desired treatment benefits, while ‘damage to the liver, kidneys or eyes’ was the most concerning risk. Factors influencing their tolerance to risks related to previously having experienced the risk and differentiation between permanent and temporary risks. A few differences were elicited between patients and caregivers. Conclusions This qualitative study provided an open forum to elicit treatment-desired benefits and acceptable risks to be established by patients themselves. These findings can inform decisions for developing new treatments and the design of clinical trials for DM1 and MM.

BackgroundFood may trigger Attention-Deficit/Hyperactivity Disorder (ADHD) symptoms. Therefore, an elimination diet (ED) might be an effective treatment for children with ADHD. However, earlier studies were criticized for the nature of the control group, potential confounders explaining the observed effects, unsatisfactory blinding, potential risks of nutritional deficiencies and unknown long term and cost-effectiveness. To address these issues, this paper describes the rationale, study design and methods of an ongoing two arm randomized controlled trial (RCT) comparing the short (5 week) and long term (1 year) effects of an elimination diet and a healthy diet compared with care as usual (CAU) in children with ADHD.MethodsA total ofN = 162 children (5–12 years) with ADHD will be randomized to either an ED or a healthy diet. A comparator arm includingN = 60 children being solely treated with CAU (e.g. medication) is used to compare the effects found in both dietary groups. The two armed RCT is performed in two youth psychiatry centers in the Netherlands, with randomization within each participating center. The primary outcome measure is response to treatment defined as a ≥ 30% reduction on an ADHD DSM-5 rating scale (SWAN) and/or on an emotion dysregulation rating scale (SDQ: dysregulation profile). This is assessed after 5 weeks of dietary treatment, after which participants continue the diet or not. Secondary outcome measures include the Disruptive Behavior Diagnostic Observational Schedule (DB-DOS), parent and teacher ratings of comorbid symptoms, cognitive assessment (e.g. executive functions), school functioning, physical measurements (e.g. weight), motor activity, sleep pattern, food consumption, nutritional quality of the diet, adherence, parental wellbeing, use of health care resources and cost-effectiveness. Assessments take place at the start of the study (T0), after five weeks (T1), four months (T2), eight months (T3) and 12 months of treatment (T4). T0, T1 and T4 assessments take place at one of the psychiatric centers. T2 and T3 assessments consist of filling out online questionnaires by the parents only.DiscussionThis RCT will likely contribute significantly to clinical practice for ADHD by offering insight into the feasibility, nutritional quality, (cost-)effectiveness and long term effects of dietary treatments for ADHD.Trial registrationwww.trialregister.nl,NTR5434. Registered at October 11th, 2015.

Samenvatting Inleiding We hebben berekend hoeveel er in 2015 aan preventie is uitgegeven. We maakten onderscheid tussen gezondheidsbescherming, gezondheidsbevordering en ziektepreventie. Bij de berekening van de uitgaven beperkten we ons tot universele, selectieve en geïndiceerde preventie. Zorggerelateerde preventie kan nauwelijks onderscheiden worden van curatieve zorg. De uitgaven aan preventie zijn berekend vanuit een maatschappelijk perspectief. Methode We hebben gevarieerde bronnen gebruikt om de uitgaven aan preventie te onderzoeken. Voor zover de kosten onderdeel waren van de uitgaven aan zorg zijn de berekeningen gebaseerd op de gegevens van het CBS. Voor het overige is gebruikgemaakt van jaarrapporten en jaarrekeningen van ministeries en andere organisaties. Ook uitgaven van consumenten, het bedrijfsleven, ngo’s en verzekeringsmaatschappijen zijn opgenomen. Resultaten In 2015 werd 12,5 miljard euro (1,8% van het bruto binnenlands product) uitgegeven aan preventie: 2,4 miljard aan ziektepreventie (18,8%), 0,6 miljard aan gezondheidsbevordering (5,2%) en 9,5 miljard aan gezondheidsbescherming (76%). Dit is 17% minder dan er in 2007 is uitgegeven, het laatste jaar dat een dergelijk onderzoek is uitgevoerd. Conclusie De uitgaven aan preventie zijn relatief laag vergeleken met de totale uitgaven aan zorg. Het grootste gedeelte gaat naar gezondheidsbescherming. In de komende jaren is er mogelijk een stijging aan uitgaven voor gezondheidsbevordering door regeringsbeleid, zoals het Nationaal Preventieakkoord.

Background: Active disinvestment of healthcare interventions (i.e. discontinuing reimbursement by means of a policy decision) has received limited public support in the past. Previous research has identified four viewpoints on active disinvestment among citizens in the Netherlands. However, it remained unclear how strong these viewpoints are supported by society, and by whom. Therefore, the current study aimed to 1) measure the support for these four viewpoints and 2) assess whether support is associated with background characteristics of citizens. Method: In an online survey, a representative sample of adult citizens in the Netherlands (n = 1794) was asked to rate their agreement with short narratives of the four viewpoints on a 7-point Likert scale. The survey also included questions on sociodemographic characteristics, health status, healthcare utilization, and opinions about responsibility and costs in the healthcare context. Logistic regression models were estimated for each viewpoint to assess the association between viewpoint support and these characteristics. Results: The support for the different viewpoints varied between 46.8% and 57.7% of the sample. Viewpoint support was associated with participants’ age, gender, educational level, financial situation, healthcare utilization, opinion on the responsibility of the government for the health of citizens, and opinion on whether the increase in healthcare expenditure and health insurance premiums is considered a problem. Conclusion: Resistance to active disinvestment may partially be explained by the consequences of disinvestment citizens anticipate experiencing themselves. Citizens considering the increase in healthcare expenditure a larger problem were more supportive of disinvestment than those considering it less of a problem.

IntroductionEar pain is the most prominent symptom of childhood acute otitis media (AOM). To control the pain and reduce reliance on antibiotics, evidence of effectiveness for alternative interventions is urgently needed. This trial aims to investigate whether analgesic ear drops added to usual care provide superior ear pain relief over usual care alone in children presenting to primary care with AOM.Methods and analysisThis is a pragmatic, two-arm, individually randomised, open, superiority trial with cost-effectiveness analysis and nested mixed-methods process evaluation in general practices in the Netherlands. We aim to recruit 300 children aged 1–6 years with a general practitioner (GP) diagnosis of AOM and ear pain. Children will be randomly allocated (ratio 1:1) to either (1) lidocaine hydrochloride 5 mg/g ear drops (Otalgan) one to two drops up to six times daily for a maximum of 7 days in addition to usual care (oral analgesics, with/without antibiotics); or (2) usual care. Parents will complete a symptom diary for 4 weeks as well as generic and disease-specific quality of life questionnaires at baseline and 4 weeks. The primary outcome is the parent-reported ear pain score (0–10) over the first 3 days. Secondary outcomes include proportion of children consuming antibiotics, oral analgesic use and overall symptom burden in the first 7 days; number of days with ear pain, number of GP reconsultations and subsequent antibiotic prescribing, adverse events, complications of AOM and cost-effectiveness during 4-week follow-up; generic and disease-specific quality of life at 4 weeks; parents’ and GPs’ views and experiences with treatment acceptability, usability and satisfaction.Ethics and disseminationThe Medical Research Ethics Committee Utrecht, the Netherlands, has approved the protocol (21-447/G-D). All parents/guardians of participants will provide written informed consent. Study results will be submitted for publication in peer-reviewed medical journals and presented at relevant (inter)national scientific meetings.Trial registrationThe Netherlands Trial Register: NL9500; date of registration: 28 May 2021. At the time of publication of the study protocol paper, we were unable to make any amendments to the trial registration record in the Netherlands Trial Register. The addition of a data sharing plan was required to adhere to the International Committee of Medical Journal Editors guidelines. The trial was therefore reregistered in ClinicalTrials.gov (NCT05651633; date of registration: 15 December 2022). This second registration is for modification purposes only and the Netherlands Trial Register record (NL9500) should be regarded as the primary trial registration.

BackgroundAround 15%–20% of children with acute otitis media present with ear discharge due to a spontaneous tear or perforation of the eardrum (AOMd). Current guidance recommends clinicians to consider oral antibiotics as first-line treatment in this condition. The opening in the eardrum however should allow topical antibiotics to enter the middle ear directly. Local administration of antibiotics does not expose children to systemic side effects and may put less selective resistance pressure on bacteria. Evidence on the effectiveness of this approach in children with AOMd is lacking.Methods and analysisA primary care-based, open, individually randomised, controlled, non-inferiority trial. The trial aims to recruit 350 children aged 6 months to 12 years with AOMd and ear pain and/or fever. Participants will be randomised to 7 days of hydrocortisone-bacitracin-colistin eardrops five drops three times daily or amoxicillin oral suspension 50 mg/kg body weight per day, divided over three doses. Parents will keep a daily diary of AOM symptoms, adverse events and complications for 2 weeks. In addition, they will record AOM recurrences, healthcare utilisation and societal costs for 3 months. The primary outcome is the proportion of children without ear pain and fever at day 3. Secondary outcomes include ear pain and fever intensity/severity; days with ear discharge; eardrum perforation at 2 weeks; adverse events during first 2 weeks; costs; and cost effectiveness at 2 weeks and 3 months. The primary analyses will be intention-to-treat and per-protocol analyses will be conducted as well.Ethics and disseminationThe medical research ethics committee Utrecht, The Netherlands has given ethical approval (17-400/G-M). Parents/guardians of participants will provide written informed consent. Study results will be submitted for publication in peer-reviewed medical journals and presented at relevant (inter)national scientific meetings.Trial registration numberThe Netherlands National Trial Register; NTR6723. Date of registration: 27 November 2017.

Long-term usage of benzodiazepines for anxiety and sleeping disorders results in a decline in effectiveness and an increase in the risk of dependence and side-effects over time. For this reason, in 2009, benzodiazepines for anxiety and sleeping disorders were disinvested (i.e. reimbursement was discontinued) in the Netherlands. Several factors contributed to disinvestment: the broad support from involved actors for disinvestment, the possibility to keep reimbursing benzodiazepines for patients groups for whom long-term use is indicated, patient groups that were not well organized, and the fact that long-term benzodiazepine use for anxiety and sleeping disorders is not medically necessary as well as ineffective. In the first year following disinvestment, the usage of benzodiazepines decreased with 15%. In subsequent years, usage stabilized, after which it decreased again.

Background Cardiometabolic diseases (CMD) are the major cause of death worldwide and are associated with a lower quality of life and high healthcare costs. To prevent a further rise in CMD and related healthcare costs, early detection and adequate management of individuals at risk could be an effective preventive strategy. The objective of this study was to determine long-term cost-effectiveness of stepwise CMD risk assessment followed by individualized treatment if indicated compared to care as usual. A computer-based simulation model was used to project long-term health benefits and cost-effectiveness, assuming the prevention program was implemented in Dutch primary care. Methods A randomized controlled trial in a primary care setting in which 1934 participants aged 45–70 years without recorded CMD or CMD risk factors participated. The intervention group was invited for stepwise CMD risk assessment through a risk score (step 1), additional risk assessment at the practice in case of increased risk (step 2) and individualized follow-up treatment if indicated (step 3). The control group was not invited for risk assessment, but completed a health questionnaire. Results of the effectiveness analysis on systolic blood pressure (− 2.26 mmHg; 95% CI − 4.01: − 0.51) and total cholesterol (− 0.15 mmol/l; 95% CI − 0.23: − 0.07) were used in this analysis. Outcome measures were the costs and benefits after 1-year follow-up and long-term (60 years) cost-effectiveness of stepwise CMD risk assessment compared to no assessment. A computer-based simulation model was used that included data on disability weights associated with age and disease outcomes related to CMD. Analyses were performed taking a healthcare perspective. Results After 1 year, the average costs in the intervention group were 260 Euro higher than in the control group and differences were mainly driven by healthcare costs. No meaningful change was found in EQ 5D-based quality of life between the intervention and control groups after 1-year follow-up (− 0.0154; 95% CI − 0.029: 0.004). After 60 years, cumulative costs of the intervention were 41.4 million Euro and 135 quality-adjusted life years (QALY) were gained. Despite improvements in blood pressure and cholesterol, the intervention was not cost-effective (ICER of 306,000 Euro/QALY after 60 years). Scenario analyses did not allow for a change in conclusions with regard to cost-effectiveness of the intervention. Conclusions Implementation of this primary care-based CMD prevention program is not cost-effective in the long term. Implementation of this program in primary care cannot be recommended. Trial registration Dutch Trial Register NTR4277, registered on 26 November 2013

Background Healthcare costs related to ESRD are well-described, but broader societal costs of ESRD are less known. This study aimed to estimate patient and family costs, including informal care costs and out-of-pocket costs, and costs due to productivity loss related to ESRD, for patients receiving dialysis and living with a kidney transplant, using a bottom-up approach. Methods A total of 655 patients were asked to complete a digital questionnaire consisting of two standardised instruments (iMCQ and iPCQ) from November 2016 through January 2017. We applied a retrospective bottom-up cost estimation by combining data from the questionnaire with unit prices from the Dutch costing manual. Results Our study sample consisted of 230 patients, of which 165 were kidney transplant recipients and 65 received dialysis. The total annual non-healthcare related costs were estimated at €8284 (SD: €14,266) for transplant recipients and €23,488 (SD: €39,434) for dialysis patients. Costs due to productivity loss contributed most to the total non-healthcare costs (66% for transplant recipients and 65% for dialysis patients), followed by informal care costs (26% resp. 29%) and out-of-pocket costs, such as medication and travel expenses (8% resp. 6%). Conclusion By exposing patient, family and productivity costs, our study revealed that dialysis and transplantation are not only costly within the healthcare system, but also incur high non-healthcare costs (18–23% resp. 35% of the total societal costs). It is important to reveal these types of non-healthcare costs in order to understand the full burden of ESRD for society and the potential impact of new therapies.

ObjectivesOur study investigates the extent to which uptake of a COVID-19 digital contact-tracing (DCT) app among the Dutch population is affected by its configurations, its societal effects, and government policies toward such an app.MethodsWe performed a discrete choice experiment among Dutch adults including 7 attributes, that is, who gets a notification, waiting time for testing, possibility for shops to refuse customers who have not installed the app, stopping condition for contact tracing, number of people unjustifiably quarantined, number of deaths prevented, and number of households with financial problems prevented. The data were analyzed by means of panel mixed logit models.ResultsThe prevention of deaths and financial problems of households had a very strong influence on the uptake of the app. Predicted app uptake rates ranged from 24% to 78% for the worst and best possible app for these societal effects. We found a strong positive relationship between people’s trust in government and people’s propensity to install the DCT app.ConclusionsThe uptake levels we find are much more volatile than the uptake levels predicted in comparable studies that did not include societal effects in their discrete choice experiments. Our finding that the societal effects are a major factor in the uptake of the DCT app results in a chicken-or-the-egg causality dilemma. That is, the societal effects of the app are severely influenced by the uptake of the app, but the uptake of the app is severely influenced by its societal effects.

Introduction: Lung cancer is the deadliest and most prevalent cancer worldwide. Lung cancer treatments have different characteristics and are associated with a range of benefits and side effects for patients. Such differences may raise uncertainty among drug developers, regulators, payers, and clinicians regarding the value of these treatment effects to patients. The value of conducting patient preference studies (using qualitative and/or quantitative methods) for benefits and side effects of different treatment options has been recognized by healthcare stakeholders, such as drug developers, regulators, health technology assessment bodies, and clinicians. However, evidence-based guidelines on how and when to conduct and use these studies in drug decision-making are lacking. As part of the Innovative Medicines Initiative PREFER project, we developed a protocol for a qualitative study that aims to understand which treatment characteristics are most important to lung cancer patients and to develop attributes and levels for inclusion in a subsequent quantitative preference survey.Methods: The study protocol specifies a four-phased approach: (i) a scoping literature review of published literature, (ii) four focus group discussions with stage III and IV Non-Small Cell Lung Cancer patients, (iii) two nominal group discussions with stage III and IV Non-Small Cell Lung Cancer patients, and (iv) multi-stakeholder discussions involving clinicians and preference experts.Discussion: This protocol outlines methodological and practical steps as to how qualitative research can be applied to identify and develop attributes and levels for inclusion in patient preference studies aiming to inform decisions across the drug life cycle. The results of this study are intended to inform a subsequent quantitative preference survey that assesses patient trade-offs regarding lung cancer treatment options. This protocol may assist researchers, drug developers, and decision-makers in designing qualitative studies to understand which treatment aspects are most valued by patients in drug development, regulation, and reimbursement.

Serena Oliveri,1 Lucilla Lanzoni,1 Serena Petrocchi,1 Rosanne Janssens,2 Elise Schoefs,2 Isabelle Huys,2 Meredith Y Smith,3,4 Ian P Smith,5 Jorien Veldwijk,5,6 G Ardine de Wit,5 Gabriella Pravettoni1 1Applied Research Division for Cognitive and Psychological Science, IEO, European Institute of Oncology IRCCS, Milan, Italy; 2Department of Pharmaceutical and Pharmacological Sciences, KU Leuven, Leuven, Belgium; 3Alexion Pharmaceuticals, Inc., Boston, MA, USA; 4University of Southern California School of Pharmacy, Los Angeles, CA, USA; 5Julius Center for Health Sciences and Primary Care, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; 6School of Health Policy & Management, Erasmus University Rotterdam, Rotterdam, NetherlandsCorrespondence: Lucilla LanzoniEuropean Institute of Oncology IRCCS, Applied Research Division for Cognitive and Psychological Science, Via Giuseppe Ripamonti, 435, Milano, 20141, ItalyTel +39 294 372054Email lucilla.lanzoni@ieo.itAbstract: The application of web-based and remotely administered surveys is becoming increasingly popular due to the fact that it offers numerous advantages over traditional paper-based or computer-based surveys completed in the presence of the researcher. However, it is unclear whether complex preference elicitation tasks administered online in highly vulnerable patient populations are also feasible. This commentary discusses opportunities and challenges of conducting quantitative patient preference studies in lung cancer patients using web-based modes of data collection. We refer to our recent experience in the context of the Patient Preference in Benefit-Risk Assessments during the Drug Life Cycle (PREFER) project. Among the main advantages were the possibility of reaching a wider and geographically distant population in a shorter timeframe while reducing the financial costs of testing, the greater flexibility offered and the reduced burden on the patients. Some limitations were also identified and should be the object of further research, including the potential lack of inclusiveness of the research, the lack of control over who is completing the survey, a poor comprehension of the study material, and ultimately a lower level of engagement with the study. Despite these limitations, experience from the PREFER project suggests that online quantitative methods for data collection may provide a valuable method to explore preferences in vulnerable patient populations beyond the COVID-19 pandemic.Keywords: discrete choice experiment, swing weighting, web-based survey, educational tool, online data collection, lung cancer

Supplemental Digital Content is available in the text., By 2030, more than 80% of Europe’s population will live in an urban environment. The urban exposome, consisting of factors such as where we live and work, where and what we eat, our social network, and what chemical and physical hazards we are exposed to, provides important targets to improve population health. The EXPANSE (EXposome Powered tools for healthy living in urbAN SEttings) project will study the impact of the urban exposome on the major contributors to Europe’s burden of disease: Cardio-Metabolic and Pulmonary Disease. EXPANSE will address one of the most pertinent questions for urban planners, policy makers, and European citizens: “How to maximize one’s health in a modern urban environment?” EXPANSE will take the next step in exposome research by (1) bringing together exposome and health data of more than 55 million adult Europeans and OMICS information for more than 2 million Europeans; (2) perform personalized exposome assessment for 5,000 individuals in five urban regions; (3) applying ultra-high-resolution mass-spectrometry to screen for chemicals in 10,000 blood samples; (4) evaluating the evolution of the exposome and health through the life course; and (5) evaluating the impact of changes in the urban exposome on the burden of cardiometabolic and pulmonary disease. EXPANSE will translate its insights and innovations into research and dissemination tools that will be openly accessible via the EXPANSE toolbox. By applying innovative ethics-by-design throughout the project, the social and ethical acceptability of these tools will be safeguarded. EXPANSE is part of the European Human Exposome Network.

Background: Advanced treatment options for non-small cell lung cancer (NSCLC) consist of immunotherapy, chemotherapy, or a combination of both. Decisions surrounding NSCLC can be considered as preference-sensitive because multiple treatments exist that vary in terms of mode of administration, treatment schedules, and benefit–risk profiles. As part of the IMI PREFER project, we developed a protocol for an online preference survey for NSCLC patients exploring differences in preferences according to patient characteristics (preference heterogeneity). Moreover, this study will evaluate and compare the use of two different preference elicitation methods, the discrete choice experiment (DCE) and the swing weighting (SW) task. Finally, the study explores how demographic (i.e., age, gender, and educational level) and clinical (i.e., cancer stage and line of treatment) information, health literacy, health locus of control, and quality of life may influence or explain patient preferences and the usefulness of a digital interactive tool in providing information on preference elicitation tasks according to patients.Methods: An online survey will be implemented with the aim to recruit 510 NSCLC patients in Belgium and Italy. Participants will be randomized 50:50 to first receive either the DCE or the SW. The survey will also collect information on participants' disease-related status, health locus of control, health literacy, quality of life, and perception of the educational tool.Discussion: This protocol outlines methodological and practical steps to quantitatively elicit and study patient preferences for NSCLC treatment alternatives. Results from this study will increase the understanding of which treatment aspects are most valued by NSCLC patients to inform decision-making in drug development, regulatory approval, and reimbursement. Methodologically, the comparison between the DCE and the SW task will be valuable to gain information on how these preference methods perform against each other in eliciting patient preferences. Overall, this protocol may assist researchers, drug developers, and decision-makers in designing quantitative patient preferences into decision-making along the medical product life cycle.

Additional file 1.

Additional file 1: Supplementary Table 1. Complementary medicine unit costs.

Additional file 2: Supplementary Table 2. Sensitivity analysis 1. Use of resources and mean costs (in €) per child (imputed, two outliers excluded).

Background To optimize the focus of future public information campaigns in The Netherlands promoting the uptake of vaccines among adults and children, we quantified the contribution of several attributes to the vaccination decision. Method We performed a discrete choice experiment (DCE) among Dutch adults including six attributes, i.e. vaccine effectiveness, vaccine-preventable burden of disease (specified in severity and frequency), accessibility of vaccination in terms of co-payment and prescription requirements, frequency of mild side-effects, population-level vaccination coverage and local vaccination coverage among family and friends. Participants answered the DCE from their own perspective (‘oneself’ group) or with regard to a vaccine decision for their youngest child (‘child’ group). The data was analysed by means of panel mixed logit models. Results We included 1547 adult participants (825 ‘oneself’ and 722 ‘child’). Vaccine effectiveness was the most important attribute in the ‘oneself’ group, followed by burden of disease (relative importance (RI) 78%) and accessibility (RI 76%). In the ‘child’ group, burden of disease was most important, but tied closely with vaccine effectiveness (RI 97%). Of less importance was the risk of mild vaccine-related side-effects and both population and local vaccination coverage. Interestingly, participants were more willing to vaccinate when uptake among the population or family and friends was high, indicating that social influence and social norms plays a role. Conclusions Vaccine effectiveness and disease severity are key attributes in vaccination decision-making for adults making a decision for themselves and for parents who decide for their children. Hence, public information campaigns for both adult and child vaccination should primarily focus on these two attributes. In addition, reinforcing social norms may be considered.

Objective: The objective of this systematic review was to review the available evidence on the disparity between willingness to accept (WTA) and willingness to pay (WTP) for healthcare goods and services. Methods: A tiered approach consisting of (1) a systematic review, (2) an aggregate data meta-analysis, and (3) an individual participant data meta-analysis was used. MEDLINE, EMBASE, Scopus, Scisearch, and Econlit were searched for articles reporting both WTA and WTP for healthcare goods and services. Individual participant data were requested from the authors of the included studies. Results: Thirteen papers, reporting WTA and WTP from 19 experiments/subgroups, were included in the review. The WTA/WTP ratios reported in these papers, varied from 0.60 to 4.01, with means of 1.73 (median 1.31) for 15 estimates of the mean and 1.58 (median 1.00) for nine estimates of the median. Individual data obtained from six papers, covering 71.2% of the subjects included in the review, yielded an unadjusted WTA/WTP ratio of 1.86 (95% confidence interval 1.52–2.28) and a WTA/WTP ratio adjusted for age, sex, and income of 1.70 (95% confidence interval 1.42–2.02). Income category and age had a statistically significant effect on the WTA/WTP ratio. The approach to handling zero WTA and WTP values has a considerable impact on the WTA/WTP ratio found. Conclusions and Implications: The results of this study imply that losses in healthcare goods and services are valued differently from gains (ratio > 1), but that the degree of disparity found depends on the method used to obtain the WTA/WTP ratio, including the approach to zero responses. Irrespective of the method used, the ratios found in our meta-analysis are smaller than the ratios found in previous meta-analyses.