Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 26, 2020; 8(24): 6364-6372
Published online Dec 26, 2020. doi: 10.12998/wjcc.v8.i24.6364
Sclerosing stromal tumor of the ovary with masculinization, Meig’s syndrome and CA125 elevation in an adolescent girl: A case report
Qian Chen, Yi-Hong Chen, Hui-Yun Tang, Yang-Mei Shen, Xin Tan
Qian Chen, Yi-Hong Chen, Hui-Yun Tang, Xin Tan, Obstetrics and Gynecology Department, West China Second University Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Qian Chen, Yi-Hong Chen, Hui-Yun Tang, Xin Tan, Key Laboratory of Birth Defects and Related Diseases of Women and Children, Sichuan University, Ministry of Education, Chengdu 610041, Sichuan Province, China
Yang-Mei Shen, Department of Pathology, West China Second University Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
Author contributions: Chen Q collected the patient’s clinical data and drafted the manuscript; Chen YH and Tang HY reviewed the literature; Shen YM contributed to analyzing and interpreting the pathological findings; Tan X designed the study and critically revised the manuscript; and all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Xin Tan, PhD, Associate Professor, Obstetrics and Gynecology Department, West China Second University Hospital, Sichuan University, No. 20 Section 3, South Renmin Road, Wuhou District, Chengdu 610041, Sichuan Province, China. tan8336@icloud.com
Received: July 2, 2020
Peer-review started: July 2, 2020
First decision: September 12, 2020
Revised: September 24, 2020
Accepted: October 20, 2020
Article in press: October 20, 2020
Published online: December 26, 2020
Processing time: 169 Days and 22.8 Hours
Abstract
BACKGROUND

Sclerosing stromal tumor (SST) is an extremely rare sex cord stromal tumor of the ovary. It was first reported and named in 1973. These tumors typically present with pelvic/abdominal pain and tenderness, a mass, and/or abnormal menses, but rarely present with masculinity in children and adolescents. Only 2 cases of these tumors have been reported in premenarchal girls, who demonstrated hormonal activity, with a history of the development of a virilizing female due to hyperandrogenism. Here, we report a case of a giant SST with obvious masculinity combined with Meig’s syndrome and CA125 elevation.

CASE SUMMARY

A 17-year-old female presented with a 7-year history of the development of masculinity and a 2-year history of amenorrhea. She had hirsutism, acne, obvious laryngeal prominence, and voice deepening. Physical examination showed a male suprapubic hair pattern and a 4.0 cm × 1.5 cm enlarged clitoris. Laboratory tests showed that the testosterone level was > 15.00 ng/mL (normal range: 0.14-0.76 ng/mL), and androstenedione level was > 10.00 ng/mL (normal range: 0.3-3.3 ng/mL). A computed tomography scan of the abdomen and pelvis was carried out and showed a large, solid and cystic, partly calcified pelvic mass in the right ovary measuring 27.1 cm × 20.0 cm × 11.0 cm, 15 cm above the umbilicus (to the level of the upper part of L1). Intraoperative findings at laparotomy revealed a large tumor arising from the right ovary. Approximately, 500 mL of pale-yellow clear liquid was found in the pelvic cavity. A right salpingo-oophorectomy was performed. Microscopic examination and immunohistochemical staining of the surgical specimen showed an SST of the ovary.

CONCLUSION

This report is remarkable as our patient was not only diagnosed with an SST of the ovary, which is extremely rare in this age group, but was the largest and most obvious reported patient with this tumor who presented with virilization. Therefore, gynecologists should be aware of this potential complication in adolescent girls with a mass in the ovary.

Keywords: Ovarian tumor, Sclerosing stromal tumor, Androgens, Adolescent, Virilization, Case report, Sex cord-stromal tumor

Core Tip: Sclerosing stromal tumor (SST) of the ovary is a rare benign subtype of a sex cord stromal tumor, which occurs early in life, with an average age of 28 years. However, we report a case of a giant SST with obvious masculinity in an adolescent girl. Gynecologists should be aware of this potential complication in adolescent girls with a mass in the ovary.