OBJECTIVES:  According to AIOM estimates, the prevalence rate of ovarian cancer is 1.3%. 8–13% of women diagnosed with epithelial OC have a germline BRCA1 or BRCA2 mutation. Specifically, the lifetime risk for OC in patients with BRCA1 mutations is 39–46% compared with 10–27% in patients with BRCA2 mutation. This study aims to estimate the cost-effectiveness ratio of a preventive testing strategy for relatives of patients with BRCA mutated cancer versus a no test strategy. METHODS:  The BRCA testing pathway was elaborated by a panel of experts. Based on this, Cost-effectiveness analysis was carried out from the Italian National Health Service perspective through a decision analytic model, within 5 years times horizon. Two alternatives were considered: 1) Preventive BRCA testing for relatives of ovarian cancer patients with positive mutation BRCA1 and BRCA2; 2) No test . Cost and effectiveness data derived from literature were discounted by 3%. Costs were analyzed from Italian payer perspective. Both deterministic and probabilistic sensitivity analyses were performed. RESULTS: Considering a cost-effectiveness threshold of €40,000/QALY, the probabilistic sensitivity analyses showed that scenario 1 was cost-effective in 97% of simulations compared with scenario 2. The ICER of scenario 1 vs 2 was dominant. CONCLUSIONS:  Preventive genetic testing in relatives of patients with ovarian cancer is cost-effective. Further research needs to be carried for applications also to breast cancer screening.