Correction: Currie et al. Reality Check 2: The Cost-Effectiveness of Policy Disallowing Body Checking in Non-Elite 13- to 14-Year-Old Ice Hockey Players. Int. J. Environ. Res. Public Health 2021, 18, 6322.

There was an error in our original publication [...].

Are We Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluations and Cost-of-Illness Studies.

BACKGROUND AND OBJECTIVES: Rare diseases have a significant impact on patients, families, the health system, and society. Measuring the socioeconomic burden is crucial to valuing interventions for rare diseases. Healthcare system costs are significant, but so are costs to other government sectors, patients, families, and society. To understand the breadth of costs captured in rare disease studies, we examined the cost categories and elements of socioeconomic burden captured in published studies. METHODS: A scoping review was conducted using five electronic databases to identify English language economic evaluations and cost-of-illness studies of interventions for rare diseases (2011-21). We mapped costs using a previously developed evidence-informed framework of socioeconomic burden costs for rare disease. RESULTS: Of 4890 studies identified, 48 economic evaluations and 22 cost-of-illness studies were included. While 18/22 cost-of-illness studies utilized a societal perspective, only 7/48 economic evaluations incorporated societal costs. Most reported cost categories related to medical costs, with medication and hospitalizations being the most common elements for both study designs. Costs borne by patients, families, and society were reported less among economic evaluations than cost-of-illness studies. These included: productivity (10% vs 77%), travel/accommodation (6% vs 68%), government benefits (4% vs 18%), and family impacts (0% vs 50%). CONCLUSIONS: Contrary to cost-of-illness analyses, most of the included economic evaluations did not account for the hidden burden of rare diseases, that is, costs borne by patients, families, and societies. Including these types of costs in future studies would provide a more comprehensive picture of the burden of disease, providing empirical data to inform how we value and make decisions regarding rare disease interventions, health policy, and resource allocation.

Developing a Framework of Cost Elements of Socioeconomic Burden of Rare Disease: A Scoping Review.

BACKGROUND AND OBJECTIVE: Rare diseases place a significant burden on patients, families, the healthcare system, and society. Evidence on the socioeconomic burden of rare disease is limited and mostly reflects diseases where treatments are available. We developed a framework encompassing recommended cost elements for studies of the socioeconomic burden of rare diseases. METHODS: A scoping review, conducted in five databases (Cochrane Library, EconLit, Embase, MEDLINE, and APA PsycINFO), identified English language publications from 2000 to 2021 presenting frameworks developed for determining, measuring or valuing costs for rare or chronic diseases. Cost elements were extracted and used to develop a literature-informed framework. Structured feedback was gathered from experts in rare diseases, health economics/health services, and policy research to revise the framework. RESULTS: Of 2990 records identified, eight papers were included and informed our preliminary framework; three focused on rare disease and five on chronic disease. Following expert input, we developed a framework consisting of nine cost categories (inpatient, outpatient, community, healthcare products/goods, productivity/education, travel/accommodation, government benefits, family impacts, and other), with several cost elements within each category. Our framework includes unique costs, added from the expert feedback, including genetic testing to inform treatment, use of private laboratories or out-of-country testing, family involvement in foundations and organizations, and advocacy costs for special access programs. CONCLUSIONS: Our work is the first to identify a comprehensive list of cost elements for rare disease for use by researchers and policy makers to fully capture socioeconomic burden. Use of the framework will increase the quality and comparability of future studies. Future work should focus on measuring and valuing these costs through onset, diagnosis, and post-diagnosis.

An Economic Evaluation of Disallowing Body Checking in 11- to 12-Year-Old Ice Hockey Leagues.

BACKGROUND: After a national policy change in 2013 disallowing body checking in Pee Wee ice hockey games, the rate of injury was reduced by 50% in Alberta. However, the effect on associated health care costs has not been examined previously. HYPOTHESIS: A national policy removing body checking in Pee Wee (ages 11-12 years) ice hockey games will reduce injury rates, as well as costs. STUDY DESIGN: Cost-effectiveness analysis alongside cohort study. LEVEL OF EVIDENCE: Level 3. METHODS: A cost-effectiveness analysis was conducted alongside a cohort study comparing rates of game injuries in Pee Wee hockey games in Alberta in a season when body checking was allowed (2011-2012) with a season when it was disallowed after a national policy change (2013-2014). The effectiveness measure was the rate of game injuries per 1000 player-hours. Costs were estimated based on associated health care use from both the publicly funded health care system and privately paid health care cost perspectives. Probabilistic sensitivity analysis was conducted using bootstrapping. RESULTS: Disallowing body checking significantly reduced the rate of game injuries (-2.21; 95% CI [-3.12, -1.31] injuries per 1000 player-hours). We found no statistically significant difference in public health care system (-$83; 95% CI [-$386, $220]) or private health care costs (-$70; 95% CI [-$198, $57]) per 1000 player-hours. The probability that the policy of disallowing body checking was dominant (with both fewer injuries and lower costs) from the perspective of the public health care system and privately paid health care was 78% and 92%, respectively. CONCLUSION: Given the significant reduction in injuries, combined with lower public health care system and private costs in the large majority of iterations in the probabilistic sensitivity analysis, our findings support the policy change disallowing body checking in ice hockey in 11- and 12-year-old ice hockey leagues.

Reality Check 2: The Cost-Effectiveness of Policy Disallowing Body Checking in Non-Elite 13- to 14-Year-Old Ice Hockey Players.

Sport-related injuries are the leading cause of injury in youth and are costly to the healthcare system. When body checking is disallowed in non-elite levels of Bantam (ages 13-14 years) ice hockey, the injury rate is reduced, but the impact on costs is unknown. This study compared rates of game injuries and costs among non-elite Bantam ice hockey leagues that disallow body checking to those that did not. Methods: An economic evaluation was conducted alongside a prospective cohort study comparing 608 players from leagues where body checking was allowed in games (Calgary/Edmonton 2014-2015, Edmonton 2015-2016) with 396 players from leagues where it was not allowed in games (Vancouver, Kelowna 2014-2015, Calgary in 2015-2016). The effectiveness measure was rate of game injuries per 1000 player-hours. Costs were estimated based on associated healthcare use within the publicly funded healthcare system as well as privately paid healthcare costs. Probabilistic sensitivity analysis was conducted using bootstrapping. Results: Disallowing body checking reduced the rate of injuries by 4.32 per 1000 player-hours (95% CI -6.92, -1.56) and reduced public and total healthcare system costs by $1556 (95% CI -$2478, -$559) and $1577 (95% CI -$2629, -$500) per 1000 player-hours, respectively. These finding were robust in over 99% of iterations in sensitivity analyses in the public healthcare and the total healthcare system perspectives. There was no statistically significant difference in privately paid healthcare costs (-$65 per 1000 player-hours (95% CI -$220, $99)). Interpretation: Disallowing body checking in non-elite 13-14-year-old ice hockey nationally would prevent injuries and reduce public healthcare costs.

Implementing economic evaluation in simulation-based medical education: challenges and opportunities.

CONTEXT: Simulation-based medical education (SBME) is now ubiquitous at all levels of medical training. Given the substantial resources needed for SBME, economic evaluation of simulation-based programmes or curricula is required to demonstrate whether improvement in trainee performance (knowledge, skills and attitudes) and health outcomes justifies the cost of investment. Current literature evaluating SBME fails to provide consistent and interpretable information on the relative costs and benefits of alternatives. CONTENT: Economic evaluation is widely applied in health care, but is relatively scarce in medical education. Therefore, in this paper, using a focus on SBME, we define economic evaluation, describe the key components, and discuss the challenges associated with conducting an economic evaluation of medical education interventions. As a way forward to the rigorous and state of the art application of economic evaluation in medical education, we outline the steps to gather the necessary information to conduct an economic evaluation of simulation-based education programmes and curricula, and describe the main approaches to conducting an economic evaluation. CONCLUSION: A properly conducted economic evaluation can help stakeholders (i.e., programme directors, policy makers and curriculum designers) to determine the optimal use of resources in selecting the modality or method of assessment in simulation. It also helps inform broader decision making about allocation of scarce resources within an educational programme, as well as between education and clinical care. Economic evaluation in medical education research is still in its infancy, and there is significant potential for state-of-the-art application of these methods in this area.

Individual Preferences for Child and Adolescent Vaccine Attributes: A Systematic Review of the Stated Preference Literature.

BACKGROUND: Discrete choice experiments are increasingly used to assess preferences for vaccines and vaccine service delivery. OBJECTIVES: To synthesize and critically assess the application of discrete choice experiments in childhood/adolescent vaccines, to describe how discrete choice experiments have been applied to understand preferences, and to evaluate the use of discrete choice experiment data to inform estimates of vaccine uptake. METHODS: We conducted a systematic review of six electronic databases. Included studies were discrete choice experiments and conjoint analyses published from 2000 to 2016 related to childhood/adolescent vaccines where respondents were parents, children/adolescents, or service providers. Validity assessment was used to assess study quality and risk of bias. RESULTS: In total, 27 articles were included, representing 21 different studies. A majority of articles were published between 2011 and 2016. Vaccines studied included human papillomavirus (24%), influenza (19%), meningococcal vaccines (14%), childhood vaccines (14%), hypothetical vaccines (10%), hepatitis B (5%), and diphtheria, tetanus, pertussis, hepatitis B, poliomyelitis, and Haemophilus influenzae type b (5%). Most studies assessed parent preferences (67%). The most common attributes were risk (24%), degree/duration of protection (21%), and cost (15%). Commonly reported outcome measures were estimates of uptake (33%), willingness-to-pay (22%), and other marginal rates of substitution (14%). Validity assessments yielded high scores overall. Areas of weakness included low response rates, inefficient experimental design, and failure to conduct formative qualitative work and a pilot of the discrete choice experiment. CONCLUSION: This is the first systematic review of childhood/adolescent vaccine-related discrete choice experiments. In future, special attention should be paid to ensuring that choice context and discrete choice experiment design are compatible to generate reliable estimates of uptake.

Alberta Provincial Pediatric EnTeric Infection TEam (APPETITE): epidemiology, emerging organisms, and economics.

BACKGROUND: Each year in Canada there are 5 million episodes of acute gastroenteritis (AGE) with up to 70% attributed to an unidentified pathogen. Moreover, 90% of individuals with AGE do not seek care when ill, thus, burden of disease estimates are limited by under-diagnosing and under-reporting. Further, little is known about the pathogens causing AGE as the majority of episodes are attributed to an "unidentified" etiology. Our team has two main objectives: 1) to improve health through enhanced enteric pathogen identification; 2) to develop economic models incorporating pathogen burden and societal preferences to inform enteric vaccine decision making. METHODS/DESIGN: This project involves multiple stages: 1) Molecular microbiology experts will participate in a modified Delphi process designed to define criteria to aid in interpreting positive molecular enteric pathogen test results. 2) Clinical data and specimens will be collected from children aged 0-18 years, with vomiting and/or diarrhea who seek medical care in emergency departments, primary care clinics and from those who contact a provincial medical advice line but who do not seek care. Samples to be collected will include stool, rectal swabs (N = 2), and an oral swab. Specimens will be tested employing 1) stool culture; 2) in-house multiplex (N = 5) viral polymerase chain reaction (PCR) panel; and 3) multi-target (N = 15) PCR commercially available array. All participants will have follow-up data collected 14 days later to enable calculation of a Modified Vesikari Scale score and a Burden of Disease Index. Specimens will also be collected from asymptomatic children during their well child vaccination visits to a provincial public health clinic. Following the completion of the initial phases, discrete choice experiments will be conducted to enable a better understanding of societal preferences for diagnostic testing and vaccine policy. All of the results obtained will be integrated into economic models. DISCUSSION: This study is collecting novel samples (e.g., oral swabs) from previously untested groups of children (e.g., those not seeking medical care) which are then undergoing extensive molecular testing to shed a new perspective on the epidemiology of AGE. The knowledge gained will provide the broadest understanding of the epidemiology of vomiting and diarrhea of children to date.

Willingness to pay for what? A note on alternative definitions of health care program benefits for contingent valuation studies.

The authors examine a number of ways in which willingness to pay (WTP) can be defined for measurement and use in a cost-benefit analysis (CBA) of a collectively funded health care program. They show how ambiguous specification of the program consequences that respondents should consider in their WTP responses can lead to problems of double counting or zero countingin a subsequent CBA. An example is whether the value of lost time from work because of poor health should be included by a CBA analyst (e.g., valued at the wage rate) as a separate cost item or whether this has already been monetized and included in respondents' WTP data. The authors highlight how differences in assumed or actual institutional structures are often ignored in measures of WTP and the consequences of this for the interpretation of WTP data.