Introduction: During pregnancy, uterine leiomyomas may cause problems and treatment typically entails uterine conservation. However, for cases of leiomyomas larger than a particular size with some clinical symptoms, enucleation should be performed. A 47-year-old female visited an obstetrics and gynecology clinic with a primary complaint of irregular bleeding. We suspected an 8.4 × 6.6 cm myoma uteri and recommended immediate surgery. During the next visit, a pregnancy test was positive and the patient requested a follow-up for her myoma uteri diagnosis. Because of a breech presentation, we performed an elective cesarean section (CS) at 38 weeks and 1 day. Methods: The patient’s myoma uteri was stable throughout the pregnancy, and after delivery, we continued to follow her up as an outpatient. Two years after the CS, the myoma uterus was 6 cm in size, and 6 months later, it had increased to 10 cm. Magnetic resonance imaging supported a diagnosis of uterine leiomyosarcoma and she underwent surgery. Results: Ultimately, she was pathologically diagnosed with uterine leiomyomas, uterine leiomyomas with bizarre nuclei, and uterine leiomyosarcoma following examination of the excised tissue. Furthermore, this case demonstrated the usefulness of cyclin E and Ki-67 as biomarkers for the malignancy of uterine mesenchymal tumors. Presently, she is being monitored for tumor recurrence and metastases on a quarterly basis. Conclusions: For detection of uterine LMS at early stage, it is important that regular follow-up of uterine leiomyoma be performed after childbirth and during pregnancy.

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