Background: Lipoleiomyomas are uncommon benign neoplasms composed of various mixtures of long intersecting bundles of bland smooth-muscle cells and mature adipocytes. These neoplasms are predominantly located in uteri and commonly occur in asymptomatic perimenopausal and postmenopausal women. Although radiologic examination is important for preoperative diagnosis and discerning the exact location of a lipoleiomyoma, the final pathologic examination confirms this diagnosis. This case of lipoleiomyoma of the uterus in a premenopausal woman is reported because because of this patient's rare presentation. Case: A 40-year-old, gravida 3, para 3, premenopausal woman presented with pain in her abdomen and dyspepsia for 45 days. Contrast-enhanced computed tomography scan results were suggestive of a possible mitotic etiology of ovarian origin. An exploratory laparotomy was performed and the neoplasm was examined histologically. Results: During the operation, the mass was found to be a large subserosal cervical fibroid growth, but the patient had healthy ovaries. The histopathology was distinctive and typical of a lipoleiomyoma. Conclusions: It is important to differentiate a lipoleiomyoma from an ovarian neoplasm that requires surgical excision. ( J GYNECOL SURG 30:24)