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Uterine leiomyoma is the most frequently occurring neoplasm of the genital tract in women; however, benign metastasizing leiomyoma (BML) is rarely seen described as a mature leiomyoma at sites distant from the uterus. To date, approximately 100 cases have been reported, with retroperitoneal BML occurring in less than ten cases. In this report, a 45-year-old asymptomatic woman who underwent a laparoscopic myomectomy six years previously was referred to the present hospital with a suspicion of recurrent uterine leiomyoma. During an exploratory laparotomy conducted in the present department, a retroperitoneal BML (6×6×4 cm³ ) was found. The postoperative pathological examination diagnosed a benign leiomyoma. Here, the authors review several current pathogenic hypotheses, the syndrome, its diagnosis, and therapy for the treatment of retroperitoneal BML. Based on this case study, the authors have demonstrated that BML can be derived from a laparoscopic myomectomy. [ABSTRACT FROM AUTHOR]