Investigation of the health economic analysis of informal care for people living with a chronic neurological disease: A systematic review and meta-analysis of the global evidence for multiple sclerosis

Multiple sclerosis (MS) is a chronic neurological disease that causes substantial health economic impacts, however, the cost of informal care for MS is often excluded from health economic analysis. As a result there is a paucity of information for decision-making. This review aims to summarise, synthesise and where appropriate meta-analyse the global evidence regarding the health economics of informal care for people with MS. The findings will provide consolidated evidence that policymakers and other stakeholders can use to inform decisions, including the development of health economics models. This review was conducted with a pre-determined study protocol (PROSPERO- CRD42023396457). Biomedical and economic databases were searched. Costs were converted to 2022 United States dollars (USD). Mean cost was calculated and pooled with a random-effects model. Subgroup analysis and meta-regression was conducted for stratified variables such as country income level and Expanded Disability Status Scale (EDSS). Of 6,306 identified studies, 61 were retained for narrative synthesis and 50 for meta-analysis. Studies were conducted in 25 countries. Cost information was collected from the person with MS, not the caregiver. 83.6% of studies used the opportunity cost method. Average monthly caregiving time was 60.1 h. Informal care costs accounted for 15% of total societal cost of MS. Pooled mean annual cost of informal care per person was USD $6,308 (95% CI $5,022-7,594). Informal care costs were USD $6,797 and $1,478 in high- and middle-income countries. Costs for mild, moderate, and severe disability of the person with MS were $1,123, $6,643, and $15,855, respectively. Informal care cost contributes considerably to MS-related costs. Despite study heterogeneity, cost of informal care increases with MS-related disability severity, time attributed to care and country income level. These results can be used to inform health economic models for reimbursement decisions for MS. Future studies regarding the health economic burden of informal care should gather the data from the informal carers themselves. •The burden of informal care is an important consideration for health economics.•Many health economics studies do not appropriately consider informal care.•We reviewed the extant literature regarding informal care for multiple sclerosis.•Informal care resource use and costs was not gathered from the carers themselves.•Informal care cost was substantial – our results can be used