This study compares adherence to breast and ovarian cancer screening recommendations among a population cohort of women at familial risk of breast and/or ovarian cancer. This cross-sectional study included 1039 first-degree female relatives without breast cancer identified from the Ontario site of the Breast Cancer Family Registry. We compared breast and ovarian cancer screening behaviors, using a telephone-administered questionnaire among three groups of women defined by their familial risk (high, moderate, and low) of breast and/or ovarian cancer. Associations between screening behaviors and familial risk were assessed using multinomial regression models adjusted by familial clustering. Women, 40-49 years of age, at moderate or high familial risk were significantly more likely to have had a screening mammogram within the past 12 months [odds ratio (OR): 2.80; 95% confidence interval (CI): 1.40-5.58], and women of less than 50 years of age were more likely to have a clinical breast examination (OR: 1.84; 95% CI: 1.02-3.31) compared with women at low familial risk. Compared with women at low or moderate familial risk, women at high familial risk were significantly more likely to have ever had a genetic test for the BRCA 1/2 genes (OR: 2.67; 95% CI: 1.76-4.05). Although the overall level of adherence among high-risk women is suboptimal in the community, women at a higher familial risk are adhering more often to cancer screening recommendations than women at a lower familial risk.