Objective:

To identify and describe cost-effectiveness studies that eva luate disease modifying therapies in the context of relapsing-remitting mul tiple sclerosis.

Method:

A systematic review of the literature was carried out by searching MEDLINE, Embase, the Cochrane Library, LILACS, the Tufts Medical Center Cost-Effectiveness Analysis Registry, the National Health Service Economic Evaluation Database and Open Grey. The search was performed in January 2018 and covered articles published between January 2010 and December 2017. The studies reviewed were payer-perspective cost-effectiveness analy ses for interferon beta-1 a, interferon beta-1 b, glatiramer acetate, teriflunomide, fingolimod, dimethyl fumarate, natalizumab, alemtuzumab and rituximab. The Quality of Health Economic Studies instrument was used to determine the quality of the studies reviewed. Risk of bias was assessed without a standar dized tool. An analysis was made of direct costs, quality-adjusted life-years and the incremental cost-effectiveness ratio. Data extraction and evaluation of information were conducted separately by each author.

Results:

Four hundred one references were found; nine studies were included. A great degree of variability was identified for several methodo logical aspects. Two studies that applied the incremental cost-effec tiveness ratio (cost) showed no first-line therapy to be cost-effective. A third study demonstrated dominance of interferon beta-1 b over placebo (USD -315,109.45) and a fourth paper showed dominance of teriflunomide over interferons and glatiramer acetate (USD -121,840.37). As regards second-line therapies, dimethyl fumarate was cost-effective in a study that compared it to glatiramer acetate and interferon beta-1a and it was dominant in another study that compared it with glatiramer ace tate (USD -158,89793) and fingolimod (USD -92,988.97). In the third line of treatment, one study showed natalizumab to be cost-effective as compared with fingolimod, and another study showed alemtuzumab to be dominant over fingolimod (USD -49,221). A third trial demonstrated alemtuzumab to be dominant over natalizumab (USD -1,656,266.07). Many of the trials have sponsorship bias. Eight of the trials received a high QHES score.

Conclusions:

The present paper shows that cost-effectiveness studies have high levels of methodological variability, some of them reaching contradictory results. As a result, it is not possible to determine which di sease-modifying therapy is really cost-effective in the context of relapsing-remitting multiple sclerosis.

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