Department of Health Services Research, Care and Public Health Research Institute (CAPHRI), Faculty of Health Medicine and Life Sciences, Maastricht University, 6200 MD, Maastricht, The Netherlands.
Institut National de Santé Publique, d'Épidémiologie Clinique et de Toxicologie (INSPECT-Lb), Beirut, Lebanon.
Pharmacoeconomics. 2021 Jul;39(7):789-807. doi: 10.1007/s40273-021-01032-7. Epub 2021 May 6.
Although the economic burden of multiple sclerosis (MS) in high-income countries (HICs) has been extensively studied, information on the costs of MS in low- and middle-income countries (LMICs) remains scarce. Moreover, no review synthesizing and assessing the costs of MS in LMICs has yet been undertaken.
Our objective was to systematically identify and review the cost of illness (COI) of MS in LMICs to critically appraise the methodologies used, compare cost estimates across countries and by level of disease severity, and examine cost drivers.
We conducted a systematic literature search for original studies in English, French, and Dutch containing prevalence or incidence-based cost data of MS in LMICs. The search was conducted in MEDLINE (Ovid), PubMed, Embase (Ovid), Cochrane Library, National Health Service Economic Evaluation Database (NHS EED), Econlit, and CINAHL (EBSCO) on July 2020 without restrictions on publication date. Recommended and validated methods were used for data extraction and analysis to make the results of the COI studies comparable. Costs were adjusted to $US, year 2019 values, using the World Bank purchasing power parity and inflated using the consumer price index.
A total of 14 studies were identified, all of which were conducted in upper-middle-income economies. Eight studies used a bottom-up approach for costing, and six used a top-down approach. Four studies used a societal perspective. The total annual cost per patient ranged between $US463 and 58,616. Costs varied across studies and countries, mainly because of differences regarding the inclusion of costs of disease-modifying therapies (DMTs), the range of cost items included, the methodological choices such as approaches used to estimate healthcare resource consumption, and the inclusion of informal care and productivity losses. Characteristics and methodologies of the included studies varied considerably, especially regarding the perspective adopted, cost data specification, and reporting of costs per severity levels. The total costs increased with greater disease severity. The cost ratios between different levels of MS severity within studies were relatively stable; costs were around 1-1.5 times higher for moderate versus mild MS and about two times higher for severe versus mild MS. MS drug costs were the main cost driver for less severe MS, whereas the proportion of direct non-medical costs and indirect costs increased with greater disease severity.
MS places a huge economic burden on healthcare systems and societies in LMICs. Methodological differences and substantial variations in terms of absolute costs were found between studies, which made comparison of studies challenging. However, the cost ratios across different levels of MS severity were similar, making comparisons between studies by disease severity feasible. Cost drivers were mainly DMTs and relapse treatments, and this was consistent across studies. Yet, the distribution of cost components varied with disease severity.
虽然高收入国家(HICs)的多发性硬化症(MS)的经济负担已经得到了广泛的研究,但关于中低收入国家(LMICs)的 MS 成本的信息仍然很少。此外,尚未对中低收入国家的 MS 成本进行综述和评估。
我们的目的是系统地确定和审查中低收入国家的 MS 疾病负担(COI),以严格评估所使用的方法,比较各国和不同疾病严重程度的成本估算,并研究成本驱动因素。
我们对中低收入国家的 MS 患病率或发病率成本数据的原始研究进行了系统的文献检索,检索使用了英语、法语和荷兰语的文献,包括 MEDLINE(Ovid)、PubMed、Embase(Ovid)、Cochrane 图书馆、国家卫生服务经济评估数据库(NHS EED)、 Econlit 和 CINAHL(EBSCO)。2020 年 7 月,没有对发表日期进行限制。使用经过推荐和验证的方法进行数据提取和分析,以确保 COI 研究结果具有可比性。使用世界银行购买力平价将成本调整为 2019 年的美元价值,并使用消费者价格指数进行通货膨胀调整。
共确定了 14 项研究,均在中上收入经济体中进行。八项研究采用自下而上的方法进行成本核算,六项研究采用自上而下的方法。四项研究采用了社会视角。每位患者的年度总成本在 463 美元至 58616 美元之间。研究之间的成本存在差异,主要是由于疾病修正疗法(DMTs)的成本、包括的成本项目范围、估计医疗资源消耗的方法等方面的差异以及对非正规护理和生产力损失的纳入。纳入研究的特征和方法差异很大,特别是在采用的视角、成本数据说明以及按严重程度水平报告成本方面。随着疾病严重程度的增加,总成本也随之增加。研究中不同 MS 严重程度之间的总成本比相对稳定,中度 MS 相对于轻度 MS 的成本约高 1-1.5 倍,重度 MS 相对于轻度 MS 的成本约高 2 倍。MS 药物成本是轻度 MS 主要的成本驱动因素,而直接非医疗成本和间接成本的比例随着疾病严重程度的增加而增加。
MS 给中低收入国家的医疗保健系统和社会带来了巨大的经济负担。研究之间存在方法学差异和绝对成本方面的显著差异,这使得研究之间的比较具有挑战性。然而,不同 MS 严重程度之间的成本比相似,因此可以按疾病严重程度进行研究之间的比较。成本驱动因素主要是 DMTs 和复发治疗,这在所有研究中都是一致的。然而,成本构成的分布随疾病严重程度而变化。