OBJECTIVES

: Through a systematic review evaluate approaches to Cost-Effectiveness Analysis (CEA) studies in the NBS field to evaluate the quality of these studies and assess consistency across them.

METHODS

: Using PRISMA guidelines we searched for all English language CEAs of blood screening in 3 databases: Medline, Embase, and Cochrane. Keywords used in the search were: cost-effectiveness, cost-benefit, cost-utility, economic evaluation, newborn screening, neonatal screening, and infant screening. A double-check by a second reviewer was done (SA). The data extracted included details about the models that have been used: perspective, outcome measure, discount rate, measurement of cost, time horizon, and other outcomes of significance. Consolidated Health Economic Evaluation Reporting Guidelines (CHEERS) were used to assess study quality.

RESULTS

: The databases yielded 2432 articles. After following PRISMA guidelines, only 33 articles were included in review (dates 1991 to 2016). These 33 studies varied widely in almost in every aspect, however, many studies were inadequate in reporting and transparency. Fewer than 10 studies followed most CHEERS guidelines. Among results were 54.5% (18 studies) used a lifetime horizon, 66.6% (22 studies) used discounting for both costs and outcomes and 51% (17 studies) used a quality-adjusted outcome (QALY or DALY).

CONCLUSIONS

: Many published CEA studies in NBS are inadequate in terms of reporting and transparency. While perfect adherence to CHEERS guidelines is a high standard (particularly as the standards were only published in 2013), the studies published in this area fall far short. While CHEERS was only published in 2013, clear direction on many of its areas of focus were available earlier in the textbook and journal literature. While CEA is a useful way of prioritizing health care interventions, those done with inadequate attention to established standards may mislead policymakers. Future CEAs in NBS should adhere to such standards.