Objective To evaluate the relationship between use of fertility medication (i.e., selective estrogen receptor [ER] modulator, gonadotropin, or other) or infertility treatment (i.e., IVF or IUI) and the risk of ovarian cancer among women with a BRCA1 or BRCA2 mutation. Design A matched case-control study of 941 pairs of BRCA1 or BRCA2 mutation carriers with and without a diagnosis of ovarian cancer. Setting Genetic clinics. Patient(s) Detailed information regarding treatment of infertility was collected from a routinely administered questionnaire. Intervention(s) None. Main Outcome Measure(s) Conditional logistic regression was used to estimate odds ratios and 95% confidence intervals associated with fertility treatment. Result(s) There was no significant relationship between the use of any fertility medication or IVF treatment (odds ratio, 0.66; 95% confidence interval 0.18–2.33) and the subsequent risk of ovarian cancer. Conclusion(s) Our findings suggest that treatment for infertility does not significantly increase the risk of ovarian cancer among women with a BRCA mutation.