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Table of Contents
CASE REPORTS
Year : 2021  |  Volume : 26  |  Issue : 3  |  Page : 208-211

Ovarian fibrothecoma—Causing menstrual disorder and hirsutism: Case report and review of literature


1 Department of Obstetrics and Gynaecology, College of Medicine, University of Lagos, Lagos University Teaching Hospital, Lagos, Nigeria
2 Department of Anatomic and Molecular Pathology, College of Medicine, University of Lagos, Lagos University Teaching Hospital, Lagos, Nigeria

Date of Submission17-Sep-2020
Date of Decision18-Dec-2020
Date of Acceptance16-Jan-2021
Date of Web Publication15-Apr-2021

Correspondence Address:
Aloy Okechukwu Ugwu
Department of Obstetrics and Gynaecology, Lagos University Teaching Hospital, Lagos
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmh.IJMH_63_20

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  Abstract 

Fibrothecomas are solid tumors of the ovary. They have been described as rare ovarian neoplasms with varied clinical manifestations. It is an uncommon tumor that originates from stromal cells accounting for 3–4% of all ovarian tumors. We present a case of a 23-year-old nulliparous undergraduate who presented to our outpatient department with secondary amenorrhea, hirsutism, and acne of one-year duration. Her symptoms worsened overtime that necessitated her presentation. Clinical examination revealed signs of hyperandrogenism with a 16-week-sized abdominopelvic mass. She had surgical excision of the mass after work-up. Her menses returned and other signs of hyperandrogenism resolved. Histopathology confirmed fibrothecoma.

Keywords: Acne, amenorrhea, fibrothecoma, hirsutism, ovarian tumor


How to cite this article:
Omisakin SI, Ugwu AO, Adebayo LA, Badmus KB. Ovarian fibrothecoma—Causing menstrual disorder and hirsutism: Case report and review of literature. Int J Med Health Dev 2021;26:208-11

How to cite this URL:
Omisakin SI, Ugwu AO, Adebayo LA, Badmus KB. Ovarian fibrothecoma—Causing menstrual disorder and hirsutism: Case report and review of literature. Int J Med Health Dev [serial online] 2021 [cited 2021 Dec 9];26:208-11. Available from: https://www.ijmhdev.com/text.asp?2021/26/3/208/313956




  Introduction Top


Sex cord-stromal tumors of the ovary originate from the dividing cells that would originally give rise to specialized gonadal stroma surrounding the oocytes, including granulosa cells, theca cells, Sertoli cells, Leydig cells, and fibroblasts.[1],[2] The tumors from these cells are a group of either benign or malignant neoplasms that arise from the sex cord (e.g. Sertoli cell tumor, granulosa cell tumor) or stromal cells (e.g. fibroma, thecoma, Leydig cell tumor) or both (Sertoli–Leydig cell tumor). sex cord stromal tumours account for <4% and 8% of benign and malignant ovarian neoplasms, respectively, and they may produce steroid hormones, especially androgen or estrogen, and hence may present with features of virilization or estrogen excess.[1],[2],[3],[4],[5],[6],[7] It is because of these bizarre clinical features that attract much fascination to sex cord stromal tumours. It is an uncommon tumor that originates from stromal cells accounting for 3–4% of all ovarian tumors.[2] Very few cases have been reported so far in Nigeria.[8]

Here, we present a case of a 23-year-old student with 1-year history of secondary amenorrhea, hirsutism, and acne due to ovarian fibrothecoma. Symptoms normalized following resection of the tumor. Ovarian fibrothecomas, although a rare tumor, have varied clinical manifestations; its therefore imperative to report this following successful treatment in order to improve quality of training.


  Case Report Top


A 23-year-old nulliparous undergraduate presented to our outpatient department with amenorrhea, hirsutism, and acne of one-year duration. There was no history suggestive of pituitary or thyroid dysfunction, no history of recurrent pelvic inflammatory disease, or prior uterine instrumentation and no associated weight loss. She has no known chronic medical condition and no prior exposure to radiation or chemotherapy. There was no change in voice, no breast atrophy, no clitoromegaly, no galactorrhea, or features of Cushing’s syndrome or acromegaly. She attained menarche at 13 years of age with a moderate flow for 4 days in a regular 30-day menstrual cycle, before the onset of present symptoms. She is sexually active.

Her physical examination revealed terminal coarse hair growth over lips, chin, upper back, and chest. Her Ferriman–Gallwey score was 13. She had acanthosis nigricans and excessive acne on the face. No abnormality was detected on respiratory and cardiovascular systems. Abdominal examination revealed no area of tenderness and no palpable organomegaly. A 16-week-sized abdominopelvic mass was noted. It was hard, firm, regular, and mobile. There was no ascites and bowel sounds were normoactive. Vaginal examination revealed a normal size anteverted uterus, 16-week-sized left adnexae mass and normal right adnexae. A diagnosis of secondary amenorrhea with an adnaexae mass was made.

Pelvic ultrasonography revealed an anteverted uterus measuring 51 mm × 36 mm × 31 mm with regular outline and homogeneous myometrial echotexture. The endometrial thickness measures 5.7 mm. No evidence of intra- or extrauterine gestation. A well-circumscribed, oval-shaped, heterogeneously hypoechoic mass measuring 62 mm × 59 mm × 57 mm was seen in the left adnexae. No calcification, necrotic area, or internal vascularity upon color Doppler was seen. The left ovary is not seen separate from the mass. The right ovary measures 26 mm × 26 mm × 10 mm, and no similar lesion was seen. The pouch of Douglas was free. Urine beta HCG was negative, and her tumor markers were within normal values: follicle-stimulating hormone 3.4 mIU/mL (0–20), prolactin 9.0 ng/mL (2–15), testosterone 3.1 ng/mL (0.2–0.95), thyroid-stimulating hormone 3.3 (0.28–4.5 mIU/mL), dehydroepiandrosterone (DHEAS) 134.6 (70.9–322 μg/dL), and luteinizing hormone (LH) 6.24 (3.2–13.5 mIU/mL). She was counseled and informed consent was obtained for surgery. Intraoperative findings were normal sized uterus, normal right ovary and tube, and normal left tube attached to the left ovarian mass measuring 14 cm × 12 cm [Figure 1]. Her immediate post-operative period was uneventful and her vital signs remained clinically stable. Her testosterone level was 0.23, 24 h following excision and her menses returned 48 h after surgery. Two weeks later, there was no coarse hair growth over her lips, chin, upper back, and chest. The histology report showed spindle-shaped cells arranged in fascicular pattern. Seen within these fascicles are clusters of oval to polyhedral cells with moderate-to-abundant vacuolated cytoplasm. There are no atypia, mitosis, or myxoid changes. There is no ovarian tissue seen [Figure 2]. A diagnosis of fibrothecoma was made.
Figure 1: Ovarian mass in a 23-year-old undergraduate

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Figure 2: (A, B and C): H&E stain showing fascicles of bland spindle cells with entrapped clusters of oval to polygonal cells with moderate-to-abundant vacuolated cytoplasm ×10, ×40, and ×100 magnifications, respectively

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  Discussion Top


This case is being reported to buttress various modes of presentation of sex cord-stromal tumors. These women can present with different symptoms ranging from abdominal or pelvic symptoms from the mass effect or as an incidental finding of adnexal mass on ultrasound. Some of the tumors may produce estrogens or androgens because of which some patients may present with signs of estrogen excess (which include abnormal uterine bleeding, endometrial hyperplasia or cancer, or, in a child, precocious puberty) or signs of androgen excess such as hirsutism, acne, alopecia (male pattern baldness), menstrual abnormalities (oligomenorrhea, amenorrhea), enlargement of the clitoris, and deepening of voice.[7] Our patient had 1-year history of amenorrhea, acne, and hirsutism which resolved following excision of the tumor. The clinical evidence of hyperperandrogenism may be attributed to the increase in testosterone production by the tumor.

Sex cord-stromal tumors are usually suspected when the patient has both an adnexal mass and endocrine effects. In such cases, the work-up will include a combination of laboratory tests (e.g. total testosterone, estradiol, tumor markers, and pelvic ultrasound scan).[3],[7],[9],[10] No specific tumor biomarker can be identified in the majority of ovarian fibromas/fibrothecomas, except for serum CA125 elevation occasionally.[9] Treatment is usually by surgery to resect and determine the histology of an ovarian mass.


  Conclusion Top


Fibrothecomas, although rare, may be an important cause of menstrual irregularity and clinical manifestations of hyperandrogenism. It is therefore necessary to increase our clinical suspicion when dealing with such conditions.

Consent

The authors certify that they have obtained all appropriate patient consent. In the consent form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understood that all her identifiable information will not be published.

Guarantor

The corresponding author will be the guarantor for this paper.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflict of interest

The authors declare that there is no conflict of interest.



 
  References Top

1.
Ano-Edward GH, Fehintola AO, Ogunlaja OA, Awotunde OT, Aaron O, Amole O, et al. A case of malignant fibrothecoma of the ovary. Ann Trop Pathol 2016:7;60-9.  Back to cited text no. 1
    
2.
Buza N, Wong S, Hui P FOXL2 mutation analysis of ovarian sex cord-stromal tumors: Genotype–phenotype correlation with diagnostic considerations. Int J Gynecol Pathol 2018;37:305-15.  Back to cited text no. 2
    
3.
Lim D, Oliva E Ovarian sex cord-stromal tumours: An update in recent molecular advances. Pathology 2018;50:178-89.  Back to cited text no. 3
    
4.
Kommoss F, Lehr HA [Sex cord-stromal tumors of the ovary: Current aspects with a focus on granulosa cell tumors, Sertoli-Leydig cell tumors, and gynandroblastomas]. Pathologe 2019;40:61-72.  Back to cited text no. 4
    
5.
Robert HY Ovarian sex cord-stromal tumours and their mimics. Pathology 2018;50:5-15.  Back to cited text no. 5
    
6.
Obeidat RA, Aleshawi AJ, Obeidat HA, Al Bashir SM A rare presentation of ovarian fibrothecoma in a middle age female: Case report. Int J Womens Health 2019;11:149-52.  Back to cited text no. 6
    
7.
Gershenson DM, Goff B, Garcia RL, Dizon DS, Chakrabati A, Vora RS Sex cord-stromal tumours of the ovary: Epidemiology, clinical features, and diagnosis in adults. Retrieved September 1 2020, https://www.uptodate.com/contents/sex-cord-stromal-tumors-of-the-ovary-granulosa-stromal-cell-tumors. Accessed January 20, 2021.  Back to cited text no. 7
    
8.
Emegoakor FC, Udealor PC, Ezenkwele EP, Nzegwu M Ovarian thecoma fibroma causing menstrual disorder & infertility: Dramatic resolution with surgery. Open J Obste Gynecol 2020;10:815-9.  Back to cited text no. 8
    
9.
Shen Y, Liang Y, Cheng X, Lu W, Xie X, Wan X Ovarian fibroma/fibrothecoma with elevated serum CA125 level: A cohort of 66 cases. Medicine (Baltimore) 2018;97:e11926.  Back to cited text no. 9
    
10.
Ramkumar S, Jyotsna VP, Mallick S, Kachhawa G, Kandasamy D, Kriplani A, et al. Bilateral thecoma presenting as premenopausal hirsutism: Laproscopic removal. Ind J Endocrinol Metab 2013;17:S662-4.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

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