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Year : 2018  |  Volume : 61  |  Issue : 2  |  Page : 261-263
Ovarian borderline mucinous tumor with squamous overgrowth: An unusual finding with review of literature

1 Department of Pathology, Kasturba Medical College, Manipal Academy of Higher Education, Udupi, Karnataka, India
2 Department of Obstetrics and Gynaecology, Kasturba Medical College, Manipal Academy of Higher Education, Udupi, Karnataka, India

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Date of Web Publication20-Apr-2018


Borderline mucinous tumor (BMT) is often associated with other entities such as dermoid cyst, Brenner tumor, and endometriosis. Squamous areas are often associated which may be a part of BMT or its associated conditions. Here, we describe squamous overgrowth in a case of intestinal type of mucinous borderline tumor in a 29-year-old uniparous female and discuss the diagnostic difficulties.

Keywords: Borderline, mucinous, overgrowth, squamous

How to cite this article:
Vasudevan G, Jaiprakash P, Guruvare S, Samanta N. Ovarian borderline mucinous tumor with squamous overgrowth: An unusual finding with review of literature. Indian J Pathol Microbiol 2018;61:261-3

How to cite this URL:
Vasudevan G, Jaiprakash P, Guruvare S, Samanta N. Ovarian borderline mucinous tumor with squamous overgrowth: An unusual finding with review of literature. Indian J Pathol Microbiol [serial online] 2018 [cited 2021 Mar 1];61:261-3. Available from: https://www.ijpmonline.org/text.asp?2018/61/2/261/230546

   Introduction Top

Borderline mucinous tumor arises from mucinous cystadenoma. They show an admixture of benign and borderline, the latter constituting at least 10% of tumor with stratified mucinous epithelium forming tufted or villiform pattern.[1] Intestinal and endocervical types are described based on lining epithelium, the latter called seromucinous or Mullerian types. Squamous overgrowth has been described in rare cases.[2]

   Case Report Top

A 29-year-old uniparous female came with a history of abdominal pain and distension for 6 months. Ultrasonography showed bilateral renal calculus with mild hydronephrosis, a hypoechoic lesion measuring 1.6 cm × 1.4 cm in the anterior wall of uterus suggestive of intramural fibroid, a well-defined hyposonolucent lesion 9 cm × 8 cm with septations in the left adnexa. CA-125 was 21.7 U/mL. Following this, computerized tomography done showed a large, well-defined hyperechoic complex solid-cystic lesion with thin and thick internal septations arising from the left adnexa with septa showing internal vascularity and nonvisualization of left ovary-likely neoplastic. A well-defined anechoic cystic lesion was arising from the right adnexa-likely simple ovarian cyst.

Preoperatively, the uterus was of normal size with left ovarian cystic mass, measuring 8 cm × 7 cm, adherent posteriorly to the fundus of uterus. The omentum and bowel appeared to be free without any deposits. Pelvic and para-aortic nodes were also not enlarged grossly.

Grossly, the hysterectomy with bilateral adnexa was received. Left-sided complex adnexal cyst was measured 7 cm × 4.5 cm and weighed 230 g. Cut section showed a multiloculated cyst filled with mucin, with thickened wall showing few papillary excrescences and focal solid areas. Microscopy showed a cystic neoplasm lined by single to focally stratified intestinal mucinous lining forming filiform and papillary structures with tufting along with extensive foci of squamous metaplasia [Figure 1], extending and forming multilayered rounded exophytic papillae [Figure 2] with invaginations into the stroma and focal neutrophilic microabscesses. The squamous cells showed intercellular bridges, with round-to-oval inconspicuous nucleoli, maintained nucleocytoplasmic ratio. However, there were no bizarre forms or evidence of invasion or even surface implants. Further, no Brenner tumor, teratoma, or endometriosis was identified, in spite of extensive sampling. Hence, a diagnosis of borderline mucinous tumor with extensive squamous overgrowth was rendered.
Figure 1: Columnar lining with areas of extensive immature squamous metaplasia (inset) with squamous lining juxtaposed with mucous cells (H and E, ×200)

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Figure 2: Papillary fronds lined by squamous epithelium lacking atypia (H and E, ×20)

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On further enquiry, the patient had a history of excision of infected right ovarian cyst, at another center. Histopathology of right cystectomy showed features of mature cystic teratoma with squamous intraepithelial neoplasia and atypical proliferation of seromucinous tumor. However, the slides were not available for review.

   Discussion Top

Fisher [1] first described the features of borderline or intermediate tumors whose clinical behavior was uncertain and did not correspond to the morphological findings. When an admixture of various epithelial types of mucinous, serous, endometrioid, and squamous lining is encountered, some authors have used the term mixed-epithelial papillary cystadenoma of borderline malignancy of Mullerian type (MEBMM).[2] In series of 37 cases of primary squamous cell carcinoma (SCC) of the ovary described by Pins et al.,[3] 11 were of pure type while 19 were associated with dermoid cyst and 7 with endometriosis. SCC associated with borderline mucinous tumor of endocervical type has also been previously described.[4]

In the present case, mucinous borderline tumor (MBT) of intestinal type had extensive areas of squamous metaplasia, with few papillae lined by mature squamous cells without any nuclear atypia. Further, these squamous cells were juxtaposed with mucinous columnar cells akin to the squamous metaplastic cells in the endocervix [Figure 2]. Multiple sections failed to reveal dysplasia, carcinoma, invasion, or foci of Brenner tumor or dermoid cyst or endometriosis, the latter three entities known to be associated with squamous overgrowth.[2]

The 2014 WHO classification also describes seromucinous tumors which contain serous and endocervical-type mucinous epithelium, along with endometrioid, indifferent, and squamous type epithelium.[5]

Although intraepithelial neoplasia can arise in ovarian cysts, as reported by Sworn et al.,[6] we did not identify any such change. There was no associated invasion as well. Intraepithelial neutrophils were however seen which has been described in MEBMM. This squamous overgrowth in MEBMM is postulated to be due the long-standing nature of the lesion.[2]

The other differential diagnoses to be considered include proliferating Brenner tumors which show foci of typical benign nests of transitional epithelium. The possibility of SCC arising in dermoid cyst, that has overgrown the dermoid, should also be considered. However, the squamous component in such cases will show obvious nuclear atypia. Transitional cell carcinomas of the ovary will also show high-grade nuclear features. In our case, extensive sampling did not reveal any other associated lesions.

In conclusion, we report an uncommon case of squamous overgrowth in MBT. We should be aware of this rare entity since it can mimic SCC due to the exuberant growth pattern of the squamous elements. The lack of nuclear features and invasion, however, help in the distinction.

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.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Hauptmann S, Friedrich K, Redline R, Avril S. Ovarian borderline tumors in the 2014 WHO classification: Evolving concepts and diagnostic criteria. Virchows Arch 2017;470:125-42.  Back to cited text no. 1
Nagai Y, Kishimoto T, Nikaido T, Nishihara K, Matsumoto T, Suzuki C, et al. Squamous predominance in mixed-epithelial papillary cystadenomas of borderline malignancy of mullerian type arising in endometriotic cysts: A study of four cases. Am J Surg Pathol 2003;27:242-7.  Back to cited text no. 2
Pins MR, Young RH, Daly WJ, Scully RE. Primary squamous cell carcinoma of the ovary. Report of 37 cases. Am J Surg Pathol 1996;20:823-33.  Back to cited text no. 3
D'Angelo E, Dadmanesh F, Pecorelli S, Prat J. Squamous cell carcinoma of the ovary arising from a mucinous cystic tumor of endocervical (müllerian) type. Int J Gynecol Pathol 2010;29:529-32.  Back to cited text no. 4
Kurman RJ, Shih IM. Seromucinous tumors of the ovary. What's in a name? Int J Gynecol Pathol 2016;35:78-81.  Back to cited text no. 5
Sworn MJ, Jones H, Letchworth AT, Herrington CS, McGee JO. Squamous intraepithelial neoplasia in an ovarian cyst, cervical intraepithelial neoplasia, and human papillomavirus. Hum Pathol 1995;26:344-7.  Back to cited text no. 6

Correspondence Address:
Padmapriya Jaiprakash
Department of Pathology, Basic Sciences Block, Kasturba Medical College, Manipal Academy of Higher Education, Madhava Nagar, Manipal, Udupi - 576 104, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_30_17

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