Background While population-based breast screening for women over the age of 50 years is a generally accepted and proven health strategy, the role of breast screening specifically among women at high risk of familial breast cancer has remained controversial. Indeed, there are very few services specifically offering a breast-screening program for women at high risk of familial breast cancer. Methods In 1999 a Familial Breast Cancer Screening Clinic (FBCSC) was established at the North Brisbane BreastScreen Queensland Service to provide a regular multimodality screening program utilizing clinical breast examination, breast ultrasound, and mammography for women at higher risk of hereditary breast cancer and with entry into the program commencing from the age of 30 years. Results Since its inception, a total of 2440 women have participated in the FBCSC. A total 7051 breast-screening examinations have been performed on these participants, with 53 breast cancers being diagnosed, including 8 in situ ductal carcinomas, 38 invasive ductal carcinomas, and 7 invasive lobular carcinomas. The mean size of the cancers was 16 mm (range = 1-45 mm), and of the 45 invasive cancers, 60% were less than or equal to 15 mm in size. The overall axillary node positive rate was 24.5% (13/53). The invasive cancer detection rate for first-round screening was 8.3 cancers per 1000 women screened, with 5.2 cancers per 1000 women detected on subsequent round screening. Conclusions The results from this service demonstrate that multimodality screening for women at high risk of familial breast cancer and including women of younger age is effective and appropriate, with very acceptable cancer detection rates and pathological cancer characteristics being observed consistent with early-stage detection. The colocated siting of this service within a BreastScreen Queensland facility has proven to be efficient and cost effective.