We present the first case of association between an ovarian endometrioma and peritoneal metastases of struma ovarii. In a 20-year-old woman, previously subjected to right adnexectomy at 12 years old for struma ovarii, during operative videolaparoscopy, we observed on left ovary the presence of endometrioma of 6 cm in diameter and on pelvic peritoneum and right lateral abdominal wall, red colored nodules of multiple sizes; one of these was visible on the diaphragm and one on the right anterior lobe of the liver. Definitive histological examination revealed endometriotic cyst, and the immunohistochemical analysis of peritoneal nodules revealed the presence of thyroid tissue.