A systematic review of recent economic evaluations of antenatal screening was conducted. Relevant studies were identified from a number of sources including computerised databases, bibliographies of economic evaluations, and searches of unpublished manuscripts. Each study identified by the literature searches was categorised on the basis of its title and abstract. Studies considered relevant to the systematic review were obtained from libraries. The methodology, results, and policy implications of studies categorised as economic evaluations upon full review were documented. A total of 566 studies were identified by the literature searches, 41 of which were categorised as economic evaluations upon full review. The economic evaluations covered a range of antenatal screening practices, aimed mainly at the prevention of infectious diseases and fetal anomalies. The review highlighted the poor methodological quality of the bulk of economic evaluations of antenatal screening. The study design, data collection methods, and analysis and interpretation of results frequently violated methodological guidelines adopted by health economists. The review also highlighted the narrow definition of benefits adopted by this body of literature, with most studies reporting outcomes in terms of cases detected, cases of particular disorders prevented or, most often, costs averted. The conclusions arrived at differed by area of antenatal screening. There appeared to be clear economic arguments in favour of some forms of antenatal screening, for example, triple test screening for Down's syndrome. Other economic evaluations pertained to specific locations, which suggests that the results may not necessarily be generalizable to different settings. For all areas of antenatal screening, an updating of published economic evaluations may be required to account for evolving economic, epidemiological, and clinical effectiveness evidence.