The patient with irregular menstruation went to our Department (Gynecological Department, the Third AffiliatedHospital, Sun Yat-sen University, Guangzhou, China) with the complaint of no pregnancy at the age of 26 in 2007, and oralcontraceptive pills were given when simple type of endometrial hyperplasia was pathologically diagnosed, and then ovulationinduction was performed. But after nearly two years from the beginning of infertility treatment, endometrial stromal sarcomawas diagnosed and radical treatment was performed for the young patient. And the relative relationship between endometrialstromal sarcoma and infertility is worth discussing.