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Synchronous benign Brenner's tumor of ovary with leiomyoma and endometrial adenocarcinoma in a postmenopausal female


1 Department of Pathology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi, India
2 Department of Obstetrics and Gynaecology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi, India

Correspondence Address:
Mohammad Jaseem Hassan,
Department of Pathology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi - 110 062
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcrt.JCRT_109_18

 > Abstract 

Brenner tumors of ovary are usually an incidental finding. It is an uncommon tumor which is seen affecting women of fifth to sixth decade. It is classified under transitional cell tumors of ovary, which includes benign, borderline, and malignant Brenner tumors and transitional cell carcinoma. These tumors have been associated with synchronous and metachronous neoplasia, most commonly other ovarian epithelial tumors such as mucinous cystadenoma. Occasionally, these tumors may be associated with endometrial hyperplasia or carcinomas which are due to hormones elaborated by the stromal component of Brenner tumor. The hormone produced is estrogen and less commonly androgens, which alters the estrogen and progesterone levels, causing hyperstimulation of endometrium. We present a case of 50-year-old postmenopausal women who presented with coexisting incidental Brenner tumor with leiomyoma and Endometrial adenocarcinoma. Only few authors have reported similar tumor occurrence in the past.

Keywords: Brenner tumor, endometrial adenocarcinoma, estrogen, ovarian tumors, synchronous tumor



How to cite this URL:
Gaur JH, Hassan MJ, Elahi AA, Khetrapal S, Khan S, Jetley S. Synchronous benign Brenner's tumor of ovary with leiomyoma and endometrial adenocarcinoma in a postmenopausal female. J Can Res Ther [Epub ahead of print] [cited 2019 Jun 16]. Available from: http://www.cancerjournal.net/preprintarticle.asp?id=251394


 > Introduction Top


Ovarian tumors represent a common neoplasia in females and comprise 30% of all tumors of female genital tract. Of the various groups of ovarian neoplasm, surface epithelial tumors are the most common comprising about 60% of all ovarian tumors. These are further categorized as serous, mucinous, endometrioid, clear cell, transitional cell, squamous cell, mixed epidermal, and undifferentiated tumor.[1] Transitional cell tumors which include Brenner, are composed of urothelium resembling epithelial components. Brenner tumor is a type of adenofibroma in which nests of transitional epithelium grow in a fibrous stroma. These are rare tumors, and constitute 2% of all ovarian tumors.[2] Benign Brenner tumor is mostly an incidental finding when oophorectomy is done for some other reason. The size is small, usually < 2 cm and most patients are asymptomatic. Brenner tumors have been seen together with other ovarian tumors and rarely with endometrial disorders ranging from endometrial atypia, hyperplasia to adenocarcinoma. Here, we report a rare case of Benign Brenner tumor, as an incidental finding in a patient with well-differentiated endometrial adenocarcinoma and leiomyoma. This is an uncommon occurrence and only occasionally reported.


 > Case Report Top


A 50-year-old postmenopausal female presented with complaints of bleeding per vaginum for 6 weeks. On examination, she was found to be obese, and her blood pressure was normal. Her hemogram showed mild anemia, other parameters were normal. On per speculum examination vulva, vagina, and cervix were found to be normal. The uterus was anteverted, mobile, and slightly bulky. Ultrasonography revealed a thickened endometrium with bilateral normal ovaries. A diagnosis of endometrial carcinoma was made on ultrasonography. The patient underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy. The specimen was sent for histopathological examination fixed in 10% normal buffered formalin. Uterus with cervix measured 10 cm × 8 cm × 4.5 cm. Right ovary measured 2.5 cm × 2.5 cm × 1.2 cm with right  Fallopian tube More Details measuring 5 cm in length, while left ovary measured 3.5 cm × 2.0 cm × 0.7 cm with left fallopian tube measuring 4.5 cm in length. Cut section of uterus showed an irregular polypoidal grayish-white growth involving almost whole of endometrial cavity with a maximum thickness of 0.8 cm [Figure 1]a. Myometrial thickness was 2.3 cm. Grossly the growth involved less than half of the myometrial thickness Furthermore, on serial sectioning of myometrium two small grayish-white leiomyoma measuring 0.5 cm and 0.3 cm were seen which shows areas of whorling [Figure 1]b. The cervix was unremarkable pearly white in color. Left ovary, on cut section, showed homogeneous firm gray-white areas. Right ovary appears unremarkable. Both fallopian tubes were unremarkable. Microscopic examination of endometrium showed well-formed glands lined by cytologically malignant columnar epithelial cells showing mild pleomorphism with scant to absent intervening stroma [Figure 2]a. The tumor involved less than half of the myometrium. A diagnosis of well-differentiated endometrial adenocarcinoma-endometrioid type (Grade 1) involving less than half of the myometrium was given. Leiomyomas found grossly were also confirmed histologically. Microscopic sections from left ovary showed nests and islands of transitional type epithelial cells showing grooved nuclei lying in a fibromatous stroma [Figure 2]b. The nests were both solid as well as few showing cystic changes. No atypia was seen. Based on these findings, a diagnosis of benign Brenner tumor was made. The other ovary and bilateral tubes were unremarkable. A final diagnosis of well-differentiated endometrial adenocarcinoma-endometrioid type (Grade 1) with Leiomyoma with incidental benign Brenner tumor of the left ovary was rendered histologically.
Figure 1: Gross examination: (a) Cut section of uterus showing an irregular polypoidal grayish-white growth involving almost whole of endometrial cavity (red arrow). Cut section of the left ovary showing homogenous firm gray-white areas (black arrow). (b) Cut section of uterus showing small grayish-white leiomyoma (green arrow)

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Figure 2: H and E staining (a) Microscopic examination of endometrium showing well-formed glands lined by cytologically malignant columnar epithelial cells showing mild pleomorphism with scant to absent intervening stroma (H and E, ×400). (b) Microscopic sections from left ovary showing nests and islands of transitional type epithelial cells lying in a fibromatous stroma with cystic change (yellow arrow) (H and E, ×400)

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 > Discussion Top


Ovarian neoplasms are an important cause of increased morbidity and mortality in females and show diverse histomorphology. The incidence of ovarian tumors has been on rise with improved medical facilities and diagnostics, the incidence in India being up to 8.4%.[3] Ovarian neoplasia incidence increases with age, with most patients presenting in premenopausal and postmenopausal age group. Patients may be asymptomatic or may present with pelvic discomfort or pain, gastrointestinal tract disturbances, ascites, and even acute abdomen if torsion occurs. Hormonally, active tumors cause abnormal uterine bleeding.

Primary ovarian tumors are broadly divided into three main categories such as surface epithelial tumors, sex cord-stromal tumors, and germ cell tumors. Transitional cell tumors are a subtype of surface epithelial tumors which include transitional cell carcinoma and Brenner tumors. The WHO categorizes Brenner tumors into three types – benign, borderline, and malignant.[1] Most common of these are benign Brenner tumor, although the overall occurrence of these benign tumors is rare constituting only 2% of all ovarian neoplasms.

Benign Brenner tumors are usually asymptomatic and an incidental finding.[4] Mostly, these tumors are small, average size is 1–2 cm, <10% of tumors reach larger size of more than 10 cm. Patients usually present in fifth to sixth decade of life with pelvic pain, abdominal discomfort, or endometrial bleeding. The patient may rarely present with Meigs syndrome when Benign Brenner tumor is associated with right-sided hydrothorax and ascites. Usually, Brenner tumor is unilateral, but bilateral tumors have also been described.[5] In our case, the ovary measured <5 cm in size and tumor was unilateral.

Brenner tumors comprise of nests and islands of oval to polygonal epithelial cells resembling urothelium and show longitudinal nuclear grooves. The cells grow is abundant dense fibroblastic stroma. The central portion of cell nests may undergo cystic degeneration. Similar findings were seen in the present case. The etiopathogenesis is unclear, although it has been accepted that Brenner tumors are derived from the surface epithelium of ovary or pelvic mesothelium which undergoes transitional cell metaplasia.[6] This has been supported by the associated tumors seen with Brenner tumors.

In about 20% of cases, associated benign ovarian tumors such as mucinous cystadenoma or serous cystadenoma, benign cystic teratoma or struma ovarii are seen. The tumors can also occur at extraovarian sites such as myometrium and omentum.[7] Brenner tumor may also be associated with endometrial abnormalities; hyperplasia and carcinoma and thus patients may present with abnormal uterine bleeding. The stromal cells are luteinized in approximately 10%–15% of Brenner tumors,[2] these cells may produce steroid hormones, mainly estrogen thus causing an imbalance between estrogen and progesterone levels. Hyperstimulation of endometrium by estrogen may be responsible for endometrial hyperplasia, atypia, and carcinoma seen with Brenner tumor.[5] This may also aid in early diagnosis as the patient become symptomatic with bleeding abnormalities.

The presence of leiomyomas has been reported with endometrial adenocarcinomas.[8],[9] This association could be attributed to the hyperestrogenic state which serves as a risk factor for the development of adenocarcinoma as well as leiomyomas.

In our case, the patient was menopausal and obese and presented with abnormal uterine bleed, found to have endometrial adenocarcinoma and small leiomyomas. This could be due to hormones produced by the synchronous Brenner tumor. Synchronous tumors are defined as two or more tumors occurring in a patient simultaneously. As many ovarian tumors are hormonally active, synchronous ovarian, and endometrial adenocarcinoma are a common occurrence.[10],[11] Typically, Type 1 endometrial adenocarcinoma occur as the presence of unopposed estrogen is an established risk factor for it.

A decision on the management of Brenner tumor depends on age and fertility status of the patient. In young patients, only simple excision with preservation of ovaries is done while in postmenopausal women, total abdominal hysterectomy with bilateral salpingo-oophorectomy is performed and is the treatment of choice as was performed in our case.[9] Prognosis of patients with Benign Brenner tumor is usually excellent.


 > Conclusion Top


Brenner tumors are mostly incidental and are found to be associated with other ovarian epithelial tumors or endometrial neoplasia. This case is a rare case of synchronous Brenner tumor with endometrial adenocarcinoma and leiomyoma in a postmenopausal female. This case is one of the few cases reported in literature highlighting the fact that Brenner tumor elaborate hormones which may be responsible for endometrial neoplasia.

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.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Available from: http://www.iarc.fr/en/publications/pdfs-online/pat-gen/bb4/bb4-chap2.pdf. [Last accessed on 2017 Jun 20].  Back to cited text no. 1
    
2.
Fletcher CD. Diagnostic Histopathology of Tumors. Philadelphia: Elsevier Health Sciences; 2013. p. 2295.  Back to cited text no. 2
    
3.
Murthy NS, Shalini S, Suman G, Pruthvish S, Mathew A. Changing trends in incidence of ovarian cancer – The Indian scenario. Asian Pac J Cancer Prev 2009;10:1025-30.  Back to cited text no. 3
    
4.
Borah T, Mahanta RK, Bora BD, Saikia S. Brenner tumor of ovary: An incidental finding. J Midlife Health 2011;2:40-1.  Back to cited text no. 4
    
5.
Indraccolo U, Cingolani N, Indraccolo SR. Bilateral brenner tumor with endometrial adenocarcinoma in a postmenopausal woman. Eur J Gynaecol Oncol 2007;28:233-4.  Back to cited text no. 5
    
6.
Arey LB. The origin and form of the Brenner tumor. Am J Obstet Gynecol 1961;81:743-51.  Back to cited text no. 6
    
7.
Hwang CS, Lee CH, Lee SJ, Kim YG, Kim A, Park DY, et al. A peculiar case report of extraovarian Brenner tumor arising in the omentum. World J Surg Oncol 2017;15:72.  Back to cited text no. 7
    
8.
Veselinova T, Gorchev G, Marinov E. Symplastic leiomyoma combined with focal adenocarcinoma of the endometrium. Akush Ginekol (Sofiia) 1996;35:47-9.  Back to cited text no. 8
    
9.
Foth D, Nawroth F, Schmidt T, Ortmann M, Römer T. Bilateral ovarian fibromas and endometrial adenocarcinoma in a postmenopausal patient with growing uterine myomas. Maturitas 2001;39:259-64.  Back to cited text no. 9
    
10.
Sharma M, Khangar B, Mallya V, Khurana N, Gupta S. Coexisting Brenner tumor and endometrial carcinoma. J Midlife Health 2017;8:89-91.  Back to cited text no. 10
    
11.
Tong SY, Lee YS, Park JS, Bae SN, Lee JM, Namkoong SE, et al. Clinical analysis of synchronous primary neoplasms of the female reproductive tract. Eur J Obstet Gynecol Reprod Biol 2008;136:78-82.  Back to cited text no. 11
    


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