OBJECTIVES:  To assess the cost and cost-utility of Bone-Anchored Hearing Aids (BAHAs) for adult and pediatric patients with bilateral deafness, mixed loss and single side deafness (SSD). METHODS:  A systematic literature review has been carried out by defining inclusion criteria according to the “PICOs” principle. In January 2016 primary economic studies published in English language have been searched in PubMed MEDLINE, Cochrane Systematic Reviews Database and Centre for Reviews and Dissemination databases. Cost data were adjusted and converted in Euro 2015. RESULTS:  43 studies have been identified and after a methodologically reproducible process, 5 economic studies have been included in the review. BAHAs was significantly more costly than convention hearing aids; for adults BAHA’s cost ranged from €6.800 in a 5-years follow up to €31.200 in lifetime horizon and for children it varied from €25.200 in a 10-years time horizon to €29.000 and €43.600 depending on one or two-staged BAHA system placement in 1 year. Cost utility studies showed that, compared with digital behind the ear hearing aid, BAHA was cost-effective with an Incremental Cost Effectiveness Ratio of €25.662 per Quality Adjusted Life Years (QALY) for adults. BAHAs was unlikely to be cost effective versus bone conduction hearing aid where benefits in terms of hearing gain and probability of using of alternative aids are similar. Incremental cost per QALY ranged from €66.500 to €142.600 for adults and from €79.300 to €170.200 for children, depending on the assumed quality of life gain and the daily use of hearing aid. CONCLUSIONS:  Few and heterogeneous studies conducted in USA and UK with the healthcare system perspective were available in the economic literature of BAHA leading to a narrative summary of results. Future research is required to provide robust cost and cost-utility estimates of BAHA, also adopting a societal perspective.