Leiomyomas can occasionally be found in the pelvic and/or abdominal parietal retroperitoneum. This rare entity is known as retroperitoneal leiomyomata. We report the case of a 46-year-old female with bilateral hydronephrosis and chronic sciatic pain caused by invasive retroperitoneal leiomyoma 3 years after hysterectomy, and a successful outcome after treatment with surgery followed by aromatase inhibitors to control pelvic residual disease. Aromatase inhibitors could be a satisfactory treatment option after surgery to control retroperitoneal leiomyoma residual disease.