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 Table of Contents  
ORIGINAL ARTICLE
Year : 2019  |  Volume : 36  |  Issue : 3  |  Page : 408-411

Ovarian seromucinous tumor: A case series of WHO newly introduced entity


Department of Pathology, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India

Date of Submission18-Apr-2019
Date of Decision27-Aug-2019
Date of Acceptance07-Nov-2019
Date of Web Publication22-Jan-2020

Correspondence Address:
Dr. E Dwivedi
Flat No 204, Upendra Apartment, Gandhi Nagar, Naria, Varansi - 221 005, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/TJOG.TJOG_30_19

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  Abstract 


Introduction: Ovarian epithelial tumors account for the majority of female ovarian neoplasms but seromucinous tumors are rare and not adequately described in the literature. The recent World Health Organization (WHO) 2014 classification of tumors of female reproductive organs introduced this new category of ovarian neoplasm as “seromucinous tumors.
Materials and Method: Sectioning of tissue followed by staining and immunohistochemistry.
Results: Four of our cases which were diagnosed as cystic lesion clinically and radiologically , on histopathological examination two of them reported as seromucinous cystadenoma and rest two as seromucinous borderline tumors (SMBT). One of the case of SMBT also showed microinvasion along with focal areas of intraepithelial carcinoma high grade and clear cell component.
Conclusion: Proper histopathological diagnosis is very important for better treatment and to reduce the use of aggressive therapies.

Keywords: Clear cell component; intraepithelial carcinoma; microinvasion; ovarian epithelial tumor; ovarian neoplasm; seromucinous borderline tumors.


How to cite this article:
Dwivedi E, Dhameja N, Lader M, Kar A G. Ovarian seromucinous tumor: A case series of WHO newly introduced entity. Trop J Obstet Gynaecol 2019;36:408-11

How to cite this URL:
Dwivedi E, Dhameja N, Lader M, Kar A G. Ovarian seromucinous tumor: A case series of WHO newly introduced entity. Trop J Obstet Gynaecol [serial online] 2019 [cited 2020 Feb 23];36:408-11. Available from: http://www.tjogonline.com/text.asp?2019/36/3/408/276434




  Introduction Top


Worldwide, ovarian cancer is the sixth most common cancer and the seventh leading cause of cancer deaths among women.[1] The recent World Health Organization (WHO) 2014 classification of Tumors of female reproductive organs introduced a new category of ovarian neoplasm designated as “seromucinous tumors” as they exhibit both serous and mucinous features.[2] These groups like other epithelial tumors include adenomas, borderline tumors, and invasive carcinomas.[3],[4] Seromucinous cystadenoma are benign cystic neoplasm with two or more Müllerian cell types, all accounting for at least 10% of the epithelial ovarian tumors (WHO 2014).[2] Seromucinous borderline tumors are characterized by papillary architecture reminiscent of serous tumors but composed of mucinous epithelium similar to that of the endocervix. These tumors are associated with endometriosis.[2],[5],[6] A few studies have reported tumors coexisting with SMBTs, such as endometrioid adenocarcinoma and squamous cell carcinoma.[7],[8]


  Case Presentation Top


Case 1

A 26-year-old female patient G3P2+0 admitted for LSCS with a complaint of lower abdominal pain and backache in the department of obstetrics. Intraoperatively, a left ovarian cyst was noted. Partial cystectomy was done and excised tissue was sent for histopathology examination. Grossly, it was an already cut open cyst measuring 5 × 2.5 cm with a wall thickness of 0.1–0.2 cm. Outer surface as well as inner surface of cyst was smooth and congested. Microscopically, sections from cyst wall was lined by variable admixture of serous and mucinous cells with a fibromatous stroma. Based on the above findings, diagnosis of Seromucinous cystadenoma was made [Figure 1].
Figure 1: H and E 20 × shows cyst wall lined by variable admixture of serous and mucinous cells with a fibromatous stroma

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Case 2

A 40-year-old female patient P2+1L2 came with a complaint of lower abdominal pain for 10-15 days in gynecology outpatient department. A contrast-enhanced computerized tomography of pelvis revealed a well-defined hypodense thick-walled cystic lesion with intermediate signal content left to the uterus, measuring about 146 × 126 mm displacing uterus toward right side likely to be ovarian cyst? endometriotic cyst? hemorrhagic cyst? Transvaginal ultrasonography Doppler is suggestive of ovarian cystadenoma. The patient underwent transabdominal hysterectomy with left adnexal cystectomy and sent for histopathological examination. Grossly, uterus showed focal areas of adenomyosis with nabothian cysts in cervix. The cystic tissue piece measured 10 × 7 cm with wall thickness of 0.4 cm. Outer surface was congested and hemorrhagic, while inner surface had much polypoidal growth with areas of hemorrhage. Microscopically, cyst showed epithelial proliferation with intracystic papillae [Figure 2]a and glands with focal areas of mucinous epithelium. Papillae and glands are lined by highly atypical cells with abundant eosinophilic to clear cytoplasm [Figure 2]b and prominent nucleoli. Dense neutrophilic infiltrate was seen within the papillary creases as well as in the epithelium. Foci of necrosis, few mural nodules [Figure 2]d, and few mitotic figures and psammoma bodies were also identified. At places, there was infiltration of the cyst wall less than 5 mm [Figure 2]c. Sheets of pigment laden macrophages and histiocytes were also seen. Immunohistochemistry was positive for CK7, ER [Figure 3]a and [Figure 3]b and negative for WT-1 [Figure 3]c, p53 [Figure 3]d. Based on above findings, diagnosis of microinvasive seromucinous borderline tumor (SMBT) with focal areas of intraepithelial carcinoma high grade and clear cell component was made.
Figure 2: (a) H and E 4 × shows epithelial proliferation with intracystic papillae. (b) H and E 40 × shows atypical cells with clear cell component. (c) H and E 40 × shows infiltration of cyst wall. (d) H and E 40 × shows mural nodule

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Figure 3: (a) CK strong positive. (b) ER positive. (c) WT1 negative. (d) P53 negative

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Case 3 and 4

A 46-year female patient P3L3 came with a complaint of menorrhagia and abnormal uterine bleeding in gynecology department. CECT of pelvis showed well defined, mixed solid cystic lesion of size 55 × 43 × 40 mm in anterioposterior (AP), transverse and craniocaudal axis, respectively, in right adnexa. The lesion showed few internal septae and enhancing solid mural component within the lesion. Similar mixed solid cystic lesion of size 44 × 40 × 28 mm in AP, transverse and craniocaudal axis, respectively, in left adnexa was suggestive of complex bilateral ovarian cysts. Transabdominal hysterectomy with bilateral salpingoopherectomy was done and sent for histopathology examination. Uterus-cervix was grossly normal with a small intramural fibroid measuring 1 × 0.5 cm. Right cystic ovary measured 7.5 × 4 × 2 cm with smooth, shiny and focal areas of congestion at outer surface. On cutting, ovary showed multiloculation with mucinous fluid in it. One of the cystic spaces showed papillary growth measuring 3 × 2 × 2 cm and rest few cystic spaces filled with solid yellowish areas measuring 1.5 × 1 × 1 cm and friable papillary growth measuring 1.5 × 1 × 0.3 cm [Figure 4]b. Left ovary measured 4 × 3 × 1 cm with three cystic spaces measuring 2 × 1 cm, 1 × 1 cm and 1 × 1 cm along with hemorrhagic and grayish-white areas. Two cystic spaces were filled with brownish jelly-like material and 3rd one with serous fluid. Microscopically, right ovary sections were lined by ciliated columnar epithelium with interspersed mucous areas along with epithelial proliferation and intraluminal broad papillae formation at places [Figure 4]c. These papillae showed neutrophilic infiltrate within them [Figure 4]a. No invasion was seen within cyst wall. Areas of hemorrhage with collection of pigment laden histiocytes was also seen. Sections from left ovary showed follicular cyst, hemorrhagic cyst, and cyst which was lined by ciliated columnar epithelium with interspersed mucous areas. Mucoid material was also present. Based on above findings diagnosis of SMBT of right ovary and seromucinous cystadenoma of left ovary was made.
Figure 4: (a) H and E 40 × papillae shows neutrophilic infiltrate within them. (b) Gross finding shows solid yellow areas with friable papillary growth. (c) H and E 4 × Epithelial proliferation with broad papillae formation at places

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  Discussion Top


In 1976, Fox and Langley first introduced the word seromucinous tumor which was composed of endocervical type mucinous epithelium and serous type of cells. Later in 1988, Rutgers and Scully described similar appearing borderline tumor into two subclasses. In 2002, Shappell et al.[6] reused the term “seromucinous tumor” as they found these neoplasms were composed of a mixture of different cell types with clinically significant differences between these two classes, hence combined into single group.[4],[9] The recent WHO classification of tumors of the female reproductive organs introduced this new class of ovarian neoplasms.[4] Morphologically, these tumors are composed of serous and endocervical type mucinous epithelium along with endometroid, indifferent, and squamous types of epithelium.[3],[4] Seromucinous cystadenoma composed of endocervical type mucinous, serous but endometroid cells and undifferentiated cells without cellular atypia. Our first case and third case (left ovary) were of same morphology. SMBTs usually arise in young women (34–44 years old) and present as unilocular or paucilocular cysts averaging 8–10 cm in diameter often with intracystic papillae.[6],[10] Our both cases (2,3) were of age 40 years and 46 years, respectively, with cystic ovary of size 10 cm and 7.5 cm showing intracystic papillae with complex papillary architecture. SMBTs may show microinvasion, intraepithelial carcinoma, and micropapillary features.[6],[7],[10],[11] Our case no 2 shows microinvasion as well as intraepithelial carcinoma of high grade. Clear cell changes are very rare finding associated with SMBT.[12] Dube et al.[7] reported a case of SMBT with endometrioid/clear cell adenofibroma. D'Angelo et al.[8] reported a case of squamous cell carcinoma that arose from a SMBT in the same ovary. In case (2), we identified clear cell component and SMBT within the same ovary. Unlike the other endometriosis-associated borderline tumors, SMBT is the most common type of tumor far exceeding benign seromucinous tumors and seromucinous carcinomas.[13] Both of our reported cases of SMBTs were associated with endometriosis. The immunohistochemical profile of atypical SMBTs reported by Vang et al.[14] reveals frequent expression of ER (100%), PR (67%), CA125 (92%), infrequent expression of WT1 (8%), and lack of expression of CK20 and CDX2, an immunostaining pattern consistent with a “müllerian” immunophenotype. A recently reported study by Taylor and McCluggage described the almost identical immunoprofile in a series of seromucinous carcinomas. Specifically, they found consistent positive expression for CK7, hormone receptors, CA125, PAX8, and CA19.9 but only a minor proportion of the tumors was positive for WT1. None of the tumors were positive for CK20 and CDX2.[13] We have used Immunohistochemistry (IHC) ER, CK7, WT-1, p53 and it was positive for ER, CK7 and negative for WT-1 and p53.


  Conclusion Top


Seromucinous cystadenoma and SMBT association with endometriosis and immunohistochemistry profile expression shows a close relationship to endometrioid and clear cell neoplasms. These tumors are associated with a good outcome. Therefore, proper histopathological diagnosis is very important for better treatment and to reduce the use of aggressive therapies.

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.
Basu P, De P, Mandal S, Ray K, Biswas J. Study of 'patterns of care' of ovarian cancer patients in a specialized cancer institute in Kolkata, Eastern India. Indian J Cancer 2009;46:28-33.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Kurman RJ, Carcangiu ML, Herrington S, Young RH. WHO classification of tumours of female reproductive organs. 4th ed. IARC; 2014.  Back to cited text no. 2
    
3.
Berenson AB, Edmonds K, Davies JP. Seromucinous cystadenoma in a true hermaphrodite: A case report. Adolesc Pediatr Gynecol 1993;6:36-8.  Back to cited text no. 3
    
4.
Kurman RJ, Shih LM. Seromucinous tumors of ovary. What's in a name? Int J Gynecol Pathol 2016;35:78-81.  Back to cited text no. 4
    
5.
Rutgers JL, Scully RE. Ovarian mixed-epithelial papillary cystadenomas of borderline malignancy of Mullerian type. A clinicopathologic analysis. Cancer 1988;61:546-54.  Back to cited text no. 5
    
6.
Shappell HW, Riopel MA, Smith Sehdev AE, Ronnett BM, Kurman RJ. Diagnostic criteria and behavior of ovarian seromucinous (endocervical-type mucinous and mixed cell-type) tumors: Atypical proliferative (borderline) tumors, intraepithelial, microinvasive, and invasive carcinomas. Am J Surg Pathol 2002;26:1529-41.  Back to cited text no. 6
    
7.
Dubé V, Roy M, Plante M, Renaud M-C, Têtu B. Mucinous ovarian tumors of mullerian-type: An analysis of 17 cases including borderline tumors and intraepithelial, microinvasive, and invasive carcinomas. Int J Gynecol Pathol 2005;24:138-46.  Back to cited text no. 7
    
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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. 8
    
9.
Lee JC, Hung YC, Yeh LS, Chang WC. Ovarian seromucinous (endocervical- type mucinous and mixed cell type) tumor: A case report. Taiwanese J Obstet Gynecol 2005;44:62-4.  Back to cited text no. 9
    
10.
Rodriguez IM, Irving JA, Prat J. Endocervical-like mucinous borderline tumors of the ovary: A clinicopathologic analysis of 31 cases. Am J Surg Pathol 2004;28:1311-8.  Back to cited text no. 10
    
11.
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. 11
    
12.
Nakamura E, Sato Y, Moriguchi S, Yamashita A, Higo T, Asada Y. Ovarian seromucinous borderline tumour and clear cell carcinoma: An unusual combination. Case Rep Obstet Gynecol 2015;2015:690891.  Back to cited text no. 12
    
13.
Taylor J, McCluggage WG. Ovarian seromucinous carcinoma: Report of a series of a newly categorized and uncommon neoplasm. Am J Surg Pathol 2015;39:983-92.  Back to cited text no. 13
    
14.
Vang R, Gown AM, Barry TS, Wheeler DT, Ronnett BM. Ovarian atypical proliferative (borderline) mucinous tumors: Gastrointestinal and seromucinous (endocervical-like) types are immunophenotypically distinctive. Int J Gynecol Pathol 2006;25:83-9.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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