Objective: To describe a case involving the cryopreservation of testis tissue retrieved from a 15-year-old male teenager with Klinefelter’s syndrome. Design: Case report. Setting: An academic medical center. Patient(s): A 15-year-old boy with Klinefelter’s syndrome. Intervention(s): Microsurgical testis sperm extraction with cryopreservation of harvested tissue. Main Outcome Measure(s): Spermatozoa within testis tissue. Result(s): Successful extraction and cryopreservation of three vials of sperm-containing testis tissue. No effect on subsequent testosterone levels. Conclusion(s): Testis tissue extraction in the adequately virilized but azoospermic young male with 47, XXY Klinefelter’s syndrome may be a strategy to preserve future biological paternity.