BackgroundDespite extensive knowledge on the functional, neurodevelopmental, behavioural and educational sequelae of preterm birth, relatively little is known about its economic consequences.ObjectiveTo systematically review evidence around the economic consequences of preterm birth for the health services, for other sectors of the economy, for families and carers, and more broadly for society.MethodsUpdating previous reviews, systematic searches of Medline, EconLit, Web of Science, the Cochrane Library, Cumulative Index to Nursing and Allied Health Literature, Embase and Scopus were performed using broad search terms, covering the literature from 1 January 2009 to 28 June 2017. Studies reporting economic consequences, published in the English language and conducted in a developed country were included. Economic consequences are presented in a descriptive manner according to study time horizon, cost category and differential denominators (live births or survivors).ResultsOf 4384 unique articles retrieved, 43 articles met the inclusion criteria. Of these, 27 reported resource use or cost estimates associated with the initial period of hospitalisation, while 26 reported resource use or costs incurred following the initial hospital discharge, 10 of which also reported resource use or costs associated with the initial period of hospitalisation. Only two studies reported resource use or costs incurred throughout the childhood years. Initial hospitalisation costs varied between $576 972 (range $111 152–$576 972) per infant born at 24 weeks’ gestation and $930 (range $930–$7114) per infant born at term (US$, 2015 prices). The review also revealed a consistent inverse association between gestational age at birth and economic costs regardless of date of publication, country of publication, underpinning study design, follow-up period, age of assessment or costing approach, and a paucity of evidence on non-healthcare costs. Several categories of economic costs, such as additional costs borne by families as a result of modifications to their everyday activities, are largely overlooked by this body of literature. Moreover, the number and coverage of economic assessments have not increased in comparison with previous review periods.ConclusionEvidence identified by this review can be used to inform clinical and budgetary service planning and act as data inputs into future economic evaluations of preventive or treatment interventions. Future research should focus particularly on valuing the economic consequences of preterm birth in adulthood.