Many benign and malignant conditions require gonadotoxic treatment regimens that may threaten a child or adolescent's future fertility. Both males and females who have undergone gonadotoxic regimens are more likely to be infertile or have difficulty getting pregnant when compared to their siblings, however the desire to parent in the future is a priority. All children who are at risk of infertility should receive a fertility preservation (FP) consultation before treatment begins. We present a case report of a prepubertal boy who was diagnosed with osteosarcoma. His initial chemotherapeutic regimen did not qualify him for a FP procedure, but when he relapsed, his subsequent therapy did. Due to his peripubertal status he attempted sperm banking, a non-experimental FP method, but was azoospermic. He ultimately achieved success with a combination of testicular tissue cryopreservation and testicular sperm extraction, experimental FP procedures. What follows is a discussion of available FP options for peripubertal males and the current state of research for male fertility restoration. Keywords: Fertility preservation, Testicular tissue cryopreservation, Testicular sperm extraction
