Purpose <p>This study aimed to characterize associations between male childhood cancer survivors’ perception of infertility risk and objective fertility status and to identify factors contributing to risk perception.</p> Methods <p>Participants were from SJLIFE, exposed to alkylating agents but not radiation, and completed a Men’s Health Questionnaire (MHQ) and semen analysis. Samples were obtained following the 2010 World Health Organization Guidelines, and classified as azoospermic (0 sperm), oligospermic (&gt; 0 and &lt; 15 million sperm/ml), or normospermic (≥ 15 million sperm/ml). Participants were classified as at minimally or high/significantly increased risk for infertility according to the Pediatric Initiative Network (PIN) criteria. Perception of infertility risk was assessed via self-report where survivors indicated the following: (1) more risk than peers without cancer history, (2) less risk, or (3) equal risk, as well as factors contextualizing perceived risk. Chi-squared tests or Fishers’ exact tests assessed differences in variables of interest.</p> Results <p>Among 238 participants (age 27 ± 6&#xa0;years, 85% NH White, 47% survivors of leukemia), 58% perceived increased infertility risk than peers without cancer history, 29% equal risk, and 13% less risk. A larger proportion of survivors with azoospermia perceived higher risk than other groups (74%) (<i>p</i> = 0.01). No differences were observed in risk perception by PIN criteria. No differences were observed by sperm concentration category or PIN criteria in factors contextualizing perceived risk.</p> Conclusions <p>Male survivors not exposed to radiation in the SJLIFE cohort are generally aware of their infertility risk. Implications for Cancer Survivors.</p> <p>Male survivors should seek continued follow-up care regarding fertility and information on risk from reputable sources.</p>