Browsing by Author "Gerber, Brittany"

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    Are we Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluation and Cost-of-Illness Studies
    (PharmacoEconomics, 2023-12) Marshall, Deborah A.; Gerber, Brittany; Lorenzetti, Diane L.; MacDonald, Karen V.; Bohach, Riley J.; Currie, Gillian R.
    Objectives: Rare diseases have a significant impact on patients, families, the health system, and society. Measuring the socioeconomic burden (SEB) 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 SEB 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 (2011-2021). We mapped costs using a previously developed evidence-informed framework of SEB 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 disease, 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.
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    Developing a Framework of Cost Elements of Socioeconomic Burden of Rare Disease: A Scoping Review
    (PharmacoEconomics, 2023-04-07) Currie, Gillian R; Gerber, Brittany; Lorenzetti, Diane; MacDonald, Karen; Benseler, Susanne M; Bernier, Francois P; Boycott, Kym M; Carias, K. Vanessa; Hamelin, Bettina; Hayeems, Robin Z; LeBlanc, Claire; Twilt, Marinka; van Rooijen, Gijs; Wong-Rieger, Durhane; Yeung, Rae S. M.; Marshall, Deborah A.
    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 and 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.
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    Horses, entertainment and sport: animal rights at the Calgary Stampede
    (2012) Gerber, Brittany; Young, Kevin M.
    In a society where questions regarding animal rights are on the rise, people are increasingly aware of the oppressive use of animals. A 'contested' view of animals has recently become the subject of sociological inquiry. Addressing the 'problematic' role of horses in modem society, this study seeks to develop an in-depth understanding of how questions of 'use' or 'abuse' have been woven into the Calgary Stampede, as illustrated by the vast Canadian media coverage annually. Specifically, this project utilized a systematic content analysis of three Canadian newspapers, combined with individual interviews with key individuals involved in the horse-use debate. Using a figurational approach, this study explores how this debate is historically situated, and how broader societal trends regarding the treatment of animals have impacted the acceptance of chuckwagon racing and rodeo as 'sports', and how various rule changes have been implemented to legitimize these events in contemporary Canadian society.
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    Uncovering the hidden socioeconomic impact of juvenile idiopathic arthritis and paving the way for other rare childhood diseases: an international, cross-disciplinary, patient-centered approach (PAVE Consortium)
    (2024-08-09) Marshall, Deborah A.; Gerber, Brittany; Currie, Gillian R.; Antón, Jordi; De Somer, Lien; Dey, Michelle; Egert, Tsipi; Egert, Yona; Henan, Lia; Klotsche, Jens; Mifsut, Laura M.; Minden, Kirsten; Normand, Christophe; Porte, David; Saurenmann, Rotraud K.; Swart, Joost F.; Uziel, Yosef; Wilson, Jennifer; Wouters, Carine; Ziv, Amit; Benseler, Susanne M.
    Abstract Background Juvenile idiopathic arthritis (JIA) refers to a heterogeneous group of rheumatic conditions in children. Novel drugs have greatly improved disease outcomes; however, outcomes are impacted by limited awareness of the importance of early diagnosis and adequate treatment, and by differences in access across health systems. As a result, patients with JIA continue to be at risk for short- and long-term morbidity, as well as impacts on virtually all aspects of life of the child and family. Main body Literature on the socioeconomic burden of JIA is largely focused on healthcare costs, and the impact of JIA on patients, families, and communities is not well understood. High quality evidence on the impact of JIA is needed to ensure that patients are receiving necessary support, timely diagnostics, and adequate treatment, and to inform decision making and resource allocation. This commentary introduces the European Joint Programme on Rare Diseases: Producing an Arthritis Value Framework with Economic Evidence: Paving the Way for Rare Childhood Diseases (PAVE) project, which will co-develop a patient-informed value framework to measure the impact of JIA on individuals and on society. With a patient-centered approach, fundamental to PAVE is the involvement of three patient advocacy organizations from Canada, Israel, and Europe, as active research partners co-designing all project phases and ensuring robust patient and family engagement. The framework will build on the findings of projects from six countries: Canada, Germany, Switzerland, Spain, Israel, and Belgium, exploring costs, outcomes (health, well-being), and unmet needs (uveitis, mental health, equity). Conclusion This unique international collaboration will combine evidence on costs (from family to societal), outcomes (clinical, patient and family outcomes), and unmet needs, to co-design and build a framework with patients and families to capture the full impact of JIA. The framework will support the development of high-quality evidence, encompassing economic and clinical considerations, unmet needs, and patient perspectives, to inform equitable resource allocation, health system planning, and quality of care better aligned with the needs of children with JIA, their families, and communities. Knowledge gained from this novel approach may pave the way forward to be applied more broadly to other rare childhood diseases.