The Cost of Cystic Fibrosis Care in Canada: A Scoping Review

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Background: Cystic fibrosis (CF) contributes a significant economic burden on individuals, health systems and society. This economic impact is highly relevant in a single-payer, publicly funded health system such as Canada. CFTR modulator therapy has been shown in large clinical trials to reduce the burden of disease in the pediatric population. Currently 17% of patients at the Alberta Children’s Hospital in Calgary, AB do not qualify for any CFTR modulator therapy, presumably resulting in an incremental cost to the system. Precision medicine tools such as organoid models may offer an opportunity for n-of-1 trials for individual patients who are currently not approved for therapy. Rationale: In order to better understand the health and economic impact of cystic fibrosis in Canada, we wish to identify direct, indirect and intangible costs of cystic fibrosis care in Alberta and Canada, and highlight the higher rate of heterogeneity amongst our southern Alberta CF population which predisposes to higher cost of care due to lack of approval to currently available therapies. Aim and Objective: This protocol describes the purpose of a scoping review which is to identify the most accurate estimation of the cost of cystic fibrosis care in Canada, and gather comprehensive data on how the costs of CF care (including direct, indirect and intangible costs) are defined and measured in Canada. Methodology: The protocol is developed using the JBI (Joanna Briggs Institute) guidance for scoping reviews and based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR) Checklist, both of which will also guide its reporting. We will search PUBMED and OVID/MEDLINE for peer-reviewed English-language publications and grey literature search of relevant databases (ProQuest Dissertation and Theses, Canadian Health Research Collection) and organizational websites (Health Canada, Government of Canada, Cystic Fibrosis Foundation of Canda, Canadian Agency for Drugs and Technology in Health (CADTH)).
Inclusion Criteria: All peer-reviewed quantitative, qualitative and mixed-methods studies conducted in Canada for patients with confirmed Cystic Fibrosis will be included. Study date range will be from 2013-2023. All studies must include reference to either the direct, indirect or intangible cost of cystic fibrosis care and management. Exclusion Criteria: Any studies conducted outside of Canada or not involving patients with cystic fibrosis, or outside the above date range will be excluded.