In 2018, the Shikoku Cancer Center established the Cancer Surveillance Clinic as a dedicated department for monitoring individuals with cancer predisposition syndromes (CPS). Notably, 30% of our patients were unaffected individuals—those without a personal history of cancer. In order to better understand the unique challenges in managing this group of patients, we have conducted a retrospective review of their clinical care. The findings revealed higher rates of clinical discontinuation and significantly lower uptake of risk-reducing salpingo-oophorectomy (RRSO) among the unaffected individuals than among those with a history of cancer. A key factor appears to be the lack of insurance coverage for CPS surveillance among unaffected individuals in Japan. However, insurance reforms alone may not be sufficient to improve engagement. The median age of the unaffected visitors was 42 years (range, 23–69 years), which is generally associated with lower cancer screening rates. This trend may reflect broader issues related to preventive health behaviors in younger populations. Many patients who discontinued care did not respond to follow-up efforts, thus suggesting a greater lack of awareness or motivation. Contributing factors may include limited public understanding of the importance of CPS surveillance, difficulty in prioritizing medical visits in the context of busy lives, and waning motivation over time. Enhancing care for this population requires not only reducing financial barriers, but also increasing public awareness of the significance of genetic risk and the role of preventive medicine in regard to CPS management.