Abstract
BackgroundDrug-resistant epilepsy negatively impacts the quality of life and is associated with increased morbidity and mortality and high costs to the healthcare system. Cannabis-based treatments may be effective in reducing seizures in this population, but whether they are cost-effective is unclear. In this systematic review, we will search for cost-effectiveness analyses involving the treatment of pediatric drug-resistant epilepsy with cannabis-based products to inform decision-making by public healthcare payers about reimbursement of such products. We will also search for cost-effectiveness analyses of other pharmacologic treatments for pediatric drug-resistant epilepsy, as well as estimates of healthcare resource use, costs, and utilities, for use in a subsequent cost-utility analysis to address this decision problem.MethodsWe will search the published and gray literature for economic evaluations of cannabis-based products and other pharmacologic treatments for pediatric drug-resistant epilepsy, as well as resource utilization and utility studies. Two independent reviewers will screen the title and abstract of each identified record and the full-text version of any study deemed potentially relevant. Study and population characteristics, the incremental cost-effectiveness ratio (ICER), as well as total costs and benefits, will be extracted, and quality will be assessed by use of the Drummond and CHEERS checklists; context-specific issues will also be considered. From model-based cost-utility and cost-effectiveness analyses, we will extract and summarize the model structure, including health states, time horizon, and cycle length. From resource utilization studies, we will extract data about the frequency of resource use (e.g., neurology visits, emergency department visits, admissions to hospital). From utility studies, we will extract the utility for each health state, the source of the preferences (e.g., child, parent, patient, general public), and the method of elicitation.DiscussionDrug-resistant epilepsy in children is associated with important costs to the healthcare system, and decision-makers require high-quality evidence on which to base reimbursement decisions. The results of this review will be useful to both decision-makers considering the decision problem of whether to reimburse cannabis-based products through public formularies and to analysts conducting studies in this area.Systematic review registrationPROSPERO no.: CRD42018099591.
Highlights
Drug-resistant epilepsy negatively impacts the quality of life and is associated with increased morbidity and mortality and high costs to the healthcare system
Pediatric epilepsy is associated with increased contacts with the healthcare system, including neurologist visits, visits to the emergency department, and admissions to hospitals [4, 5], and children with uncontrolled seizures use more health resources than children with controlled seizures, with costs correlated with epilepsy severity [4, 6]
Variation in seizure types, there is no standard of care for treatment of pediatric drug-resistant epilepsy, with treatment dependent on the nature of a child’s individual disease, which drugs are available in a given jurisdiction, and local practice [8]
Summary
This systematic review protocol has been registered in PROSPERO (CRD42018099591) and follows the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Protocols (PRISMA-P) statement (Additional file 1) [16]. Study designs: Full economic evaluations (i.e., cost-utility analysis, cost-effectiveness analysis, cost-benefit analysis, cost-minimization analysis) and health technology assessments that include a full economic evaluation Both trial-based and model-based economic evaluations will be eligible for inclusion. Study designs: Full economic evaluations and health technology assessments that involve model-based cost-utility or cost-effectiveness evaluations. Research aim 2: Model structure (e.g., description of the included health states, horizon, cycle length), model inputs (e.g., resource use [e.g., number of neurologist visits, admission to hospital, visits to the emergency department] and utility values). We will extract study characteristics (e.g., study design, location, decision problem, perspective [i.e., patient, hospital, healthcare, society], funding source), as well as population characteristics (e.g., epilepsy syndrome, age, comorbidities, setting or context) and details about the interventions and comparators. Data will be reported separately by drug-resistant epilepsy syndrome and by seizure status (e.g., controlled, uncontrolled)
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