Document Type : Case Report

Authors

• Fatemeh Amiri ^{1, 2}
• Maliheh Arab ²
• Masoomeh Raoufi ³
• Azin Kheradmand ⁴
• Behnaz Ghavami ⁵
• Behnaz Nouri ⁶

¹ Department of Obstetrics and Gynecology, Afzalipour School of Medicine, Kerman University of Medical Sciences, Kerman, Iran

² Department of Gynecology-Oncology, Faculty of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran

³ Department of Radiology, Imam Hossein, Faculty of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran

⁴ Department of Pathology, Faculty of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran

⁵ Department of Minimal Access Surgery, Faculty of Medicine, Tehran University of Medical Sciences, Tehran, Iran

⁶ Department of Obstetrics and Gynecology, Faculty of Medicine, University of Shahid Beheshti, Shohadaye Tajrish Hospital, Tehran, Iran

Abstract

 Background: Uterine tumors resembling ovarian sex cord tumors (UTROSCTs) belong to the miscellaneous mesenchymal category of uterine tumors. They typically present in middle-aged individuals, with occurrences in younger patients being rare. In the past, the primary treatment was hysterectomy, but recently, young patients, aiming to preserve fertility, have undergone fertility-preserving treatments such as tumor resection. This article presents a rare case of uterine UTROSCT in a 22-year-old female treated by local resection, under follow-up.
Case Report: A 22-year-old virgin female presented with a complaint of abnormal uterine bleeding (AUB) for the past 18 months. She underwent the initial surgical approach, which involved an incomplete hysteroscopic resection of the mass. Following this procedure, she was referred to our center after the final pathological report. Imaging revealed a uterine mass again. The patient underwent fertility-preserving surgery with laparotomy and resection of the mass, guided by hysteroscopy. Adjuvant therapy was not administered, and she has been under follow-up. Care without recurrence has been maintained for 10 months until now.
Discussion: We have performed searches using the keyword “UTROSCT” in databases including PubMed/MEDLINE, ScienceDirect, and Web of Science with a focus on English articles on under 40 years old patients published in the last decade. Among nine cases reported in scientific texts in patients under the age of 40 years, the median age was 33 years. AUB was the most common initial symptom. The tumor location was predominantly intrauterine in eight cases, including our patient, while in one case, the tumor was located in the cervix. The reported median tumor size in 6 cases was 4 centimeters, with our patient’s tumor measuring 3.5 cm. In 5 cases, for whom immunohistochemistry (IHC) was performed, the calretinin marker was positive, consistent with our patient’s case. Regarding treatment, hysterectomy with and without bilateral salpingo-oophorectomy (BSO) was performed in cases whose fertility preservation was not a concern. In our patients and cases with a focus on fertility preservation, tumor resection was performed. One reported case received adjuvant therapy after the initial surgical treatment, while our patient did not receive adjuvant treatment.
Conclusion: UTROSCT is a rare tumor, even more uncommon in reproductive age groups. Therefore, the outcomes of fertility-preserving treatments in young patients remain uncertain. Additionally, there is no definitive consensus on adjuvant therapy, and decisions should be made on a case-by-case basis. Further collection of a larger patient cohort and longer follow-up periods could provide valuable insights.

Keywords

• UTROSCT
• Young woman
• Fertility sparing surgery

Main Subjects

• Oncology