Successful monozygotic twin pregnancy fathered by a male 46,XY true hermaphrodite

This report presents an unusual case of absolute non-obstructive azoospermia revealed to be a male 46,XY true hermaphrodite who was successfully treated to father healthy monozygotic twins. A 27-year-old infertile male with non-obstructive azoospermia previously underwent an unsuccessful testicular sperm extraction procedure and refused donor sperm insemination. Revising the patient's old records revealed that he was born with ambiguous genitalia. He had a 46,XY karyotype and was raised as a male. During childhood he underwent ambiguous genitalia reconstruction, right orchiopexy and left salpingo-oophorectomy that revealed a gonadoblastoma. A new treatment was employed performing testicular fine needle aspiration leading successfully to a monozygotic twin pregnancy. As far as is known, this is the first reported case of healthy twins fathered by a male 46,XY true hermaphrodite.