Background: The presence of an ''ectopic ovary'' is rare, and the diagnosis can be difficult to establish preoperatively. Histologic confirmation is usually necessary. There has not been any case reported so far of a true ectopic ovarian leiomyoma as a cause of chronic pelvic pain. Such a case is reported in this article. Case: A 32-year-old woman presented with an incidental finding of a solid pelvic mass that had been diagnosed with a ultrasound (US) scan during a miscarriage that she had. During follow-up, she reported having symptoms of pelvic pain and deep dyspareunia. A repeat US scan showed no change in the mass that appeared to be arising from, or abutting, the posterior wall of her uterus, suggestive of a fibroid growth. Standard serum tumor markers were all normal. Magnetic resonance imaging (MRI) initially showed on only a hemorrhagic ovarian cyst. However, an initial diagnostic laparoscopy revealed a normal left ovary with a hemorrhagic cyst and a normal right ovary. However, there was also a separate solid, fibroidlike mass in her recto-uterine pouch of Douglas. Because her symptoms were persistent, a second laparoscopy was performed 10 months later, and the fibroid mass was resected. Surprisingly, the histology showed features of an ovarian leiomyoma that was considered to be ectopic in origin, as it was separate from both ovaries and was not attached to any of the adnexal structures or uterus. Results: The patient had an uneventful recovery and was completely symptomfree on follow-up. Conclusions: To the current authors' knowledge, this is the first case of a true ectopic ovarian leiomyoma reported. Successful management was achieved with minimally invasive surgery. ( J GYNECOL SURG 29:222)