Leiomyoma is a benign mesenchymal tumor that develops from smooth muscle cells. It can present in various histological variants. Leiomyoma with bizarre nuclei is an infrequent variant of uterine smooth muscle neoplasm. It is characterized by focally or diffusely distributed bizarre cells on the background of a typical leiomyoma. These bizarre cells are large, multinucleated, or multilobulated and have an eosinophilic cytoplasm. Even though leiomyomas with bizarre nuclei display benign clinical behavior, their differential diagnosis from leiomyosarcoma can sometimes be difficult. Leiomyoma has been described most commonly in the uterus. There is no case of leiomyoma originating from paratubal cysts described in the literature. In this article, we present a rare case of leiomyoma with bizarre nuclei originating from a paratubal cyst.