Leydig cell hyperplasia (LCH) is a rare cause of hyperandrogenism that has been described only in case reports. The cases presented herein contrast the traditional presentation of LCH with an affected asymptomatic individual. The first case involves a 74-year-old woman presenting with symptomatic hyperandrogenism, whose symptoms resolved after bilateral salpingo-oophorectomy (BSO). The second patient presented with postmenopausal bleeding and an abdominal mass. Following total abdominal hysterectomy (TAH) and BSO, pathology showed ovarian LCH with concomitant endometrial cancer. The diagnosis of LCH is complex and requires careful investigation of many differential diagnoses. Incidentally discovered LCH may shed light on evolution and disease progression. Cases of LCH found in the setting of endometrial pathology may have implications on other states of testosterone excess.