Purpose: Frozen-thawed embryo transfer achieves superior pregnancy and miscarriage rates compared with those achieved using fresh embryos, and there is no difference in fertilization rate using fresh versus frozen-thawed sperm collected by microdissection testicular sperm extraction (MD-TESE) when intracytoplasmic sperm injection (ICSI) is performed. However, there are few reports on the miscarriage rate using fresh versus frozen-thawed testicular sperm collected by MD-TESE. Materials and Methods: The present patient had been unable to conceive because of her husband’s azoospermia. Sperm was successfully collected via MD-TESE. ICSI using fresh or frozenthawed sperm was performed nine times. Results: Two fresh sperm ICSIs resulted in the delivery of two healthy babies. Seven frozen-thawed sperm ICSIs resulted in four pregnancies; however, three of these miscarried, and only one resulted in the delivery of a healthy baby. Conclusion: ICSI using fresh sperm decreased the rate of miscarriage compared with ICSI using frozen-thawed sperm in this patient.