OBJECTIVES: We conducted a systematic literature review to evaluate healthcare resource utilization (HCRU) and costs associated with very preterm birth (<32 weeks’ gestational age [wGA]) in North America (NA) and Europe. METHODS: Searches were conducted systematically in indexed literature databases for English-language articles published from January 2011 to July 2016. Eligible studies reported results on the economic burden (HCRU/costs) of very preterm birth in NA or Europe. RESULTS: Twenty-eight studies from NA (20 US, 8 Canada) and 20 from Europe (16 countries) were included. The majority reported HCRU/costs during the neonatal/infancy period (age <1y); 5 studies in NA and 10 in Europe reported follow-up at ≥1y, of which 1 in NA and 4 in Europe reported follow-up at ≥5y. Most studies reported inpatient or drug utilization/costs, whereas limited data were found on outpatient HCRU/costs. Total direct costs (reported in 6 studies; 4 NA, 2 Europe) among very preterm infants were generally high, particularly for those with lower GA at birth (e.g. $67,467 for children born at <28 wGA [extremely preterm], $54,554 at 28–32 wGA, $10,010 at 33–36 wGA, from birth–10y, in a Canadian study) and surgery ($196,196 vs $104,577, for infants with patent ductus arteriosus with and without surgical ligation, respectively, from birth to discharge, in a US study). Presence of chronic lung disease and congenital heart disease were also shown to increase inpatient costs from birth to discharge (2 US studies). Only 1 study in each of NA and Europe reported indirect costs, limiting conclusions that can be drawn for such outcomes. CONCLUSIONS: HCRU and costs associated with very preterm birth are substantial, and likely increase with lower GA at birth, comorbidities or need for surgery. Evidence on indirect costs and long-term economic consequences of very preterm birth is limited, with few studies reporting HCRU/costs beyond early childhood.