LGCmain
Indian Journal of Pathology and Microbiology
Home About us Instructions Submission Subscribe Advertise Contact e-Alerts Ahead Of Print Login 
Users Online: 5239
Print this page  Email this page Bookmark this page Small font sizeDefault font sizeIncrease font size


 
  Table of Contents    
CASE REPORT  
Year : 2013  |  Volume : 56  |  Issue : 1  |  Page : 51-53
Mesonephric adenocarcinoma (endometrioid type) of endocervix with diffuse mesonephric hyperplasia involving cervical wall and myometrium: An unusual case report


1 Department of Pathology, Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India
2 Department of Gynecologic Oncology, Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India

Click here for correspondence address and email

Date of Web Publication6-Aug-2013
 

   Abstract 

Malignant mesonephric tumors are rare variants of cervical adenocarcinoma, derived from remnants of mesonephric ducts and are associated with mesonephric remnants and/or mesonephric hyperplasia. Few cases have been described in literature. We report an unusual case of cervical mesonephric adenocarcinoma of endometrioid type with squamous morules in association with diffuse mesonephric hyperplasia involving the cervical walls and extending into the myometrium.

Keywords: Adenocarcinoma, endocervix, hyperplasia, mesonephric tumor

How to cite this article:
Menon S, Kathuria K, Deodhar K, Kerkar R. Mesonephric adenocarcinoma (endometrioid type) of endocervix with diffuse mesonephric hyperplasia involving cervical wall and myometrium: An unusual case report. Indian J Pathol Microbiol 2013;56:51-3

How to cite this URL:
Menon S, Kathuria K, Deodhar K, Kerkar R. Mesonephric adenocarcinoma (endometrioid type) of endocervix with diffuse mesonephric hyperplasia involving cervical wall and myometrium: An unusual case report. Indian J Pathol Microbiol [serial online] 2013 [cited 2020 Feb 28];56:51-3. Available from: http://www.ijpmonline.org/text.asp?2013/56/1/51/116150



   Introduction Top


Mesonephric duct remnants are found in - 1-22% of adult uterine cervices, mostly in the lateral walls. [1],[2] However, hyperplasia of these remnants is rare and adenocarcinomas arising from these are even rarer. Mesonephric hyperplasia is further classified as lobular mesonephric hyperplasia, diffuse mesonephric hyperplasia, and mesonephric ductal hyperplasia. [2] These categories have no clinical significance, but awareness of their features among pathologists is important to prevent their potential misdiagnosis as in situ or invasive adenocarcinoma. Mesonephric adenocarcinomas are even rarer and arise in the vicinity of mesonephric duct remnants in most cases. They may morphologically be misdiagnosed as other varieties of adenocarcinomas and immunohistochemical evaluation may be necessary to delineate these tumors. [3] These tumors are likely to have a more aggressive clinical course. [3],[4]


   Case Report Top


A 65-year-old female presented with post-menopausal bleeding of 6 months duration. On per vaginal examination, the patient had third-degree uterine prolapse and the cervix was hard with a 3 × 3 cm cervical mass. Bilateral parametria were free. On magnetic resonance imaging (MRI), a mass measuring 3.4 × 2.8 × 2.1 cm was seen protruding through the vaginal introitus, associated with third-degree uterine prolapse and cystocoele. Radiologically, endometrium, myometrium, and bilateral parametria were unremarkable. In addition, bilateral multiloculated cystic adnexal masses were identified, the left measuring 6.4 × 4.5 × 4.1 cm and the right measuring 6.5 × 5.8 × 4.5 cm. There was no significant lymphadenopathy.

Biopsy from the cervical mass revealed an adenocarcinoma with squamous morules and was reported as endometrioid adenocarcinoma, International Federation of Gynecology and Obstetrics (FIGO) grade I. The patient underwent a hysterectomy with bilateral salphigo-oopherectomy and pelvic lymph node dissection. On microscopy, the cervical mass was confirmed to be an adenocarcinoma with tubulo-glandular pattern and conspicuous squamous morules resembling an endometrioid adenocarcinoma. The cervical stroma just adjacent to the tumor revealed diffuse proliferation of mesonephric remnants in the form of small glands and tubules. These hyperplastic mesonephric tubules were seen extending into the myometrium; however, these were cytologically benign without any atypia and/or mitosis, with few of them containing intraluminal secretions [Figure 1]a-d. The tumor invaded more than half the thickness of cervical stroma (Stage IB1) and the benign tubules also extended deep into the wall of cervix. Stromal cells around adenocarcinoma did not show any atypia. There were no areas of necrosis within the tumor and no lymphovascular emboli were noted. On immunohistochemistry, both the mesonephric hyperplasia and adenocarcinoma showed immunoreactivity for cytokeratin (CK) 7. In addition, both revealed focal luminal immunostaining for CD10. The adenocarcinoma component was negative for CK20, estrogen receptor (ER), progesterone receptor (PR), and carcinoembryonic antigen (CEA) [Figure 2]a-d. Vimentin staining was seen in the hyperplastic mesonephric remnants and was negative in the adenocarcinoma. Based on the presence of these morphological and immunohistochemical features, a diagnosis of mesonephric adenocarcinoma arising on a background of mesonephric hyperplasia was rendered.
Figure 1: (a) Mesonephric adenocarcinoma (thick arrow) adjacent to hyperplasti c mesonephric remnants (thin arrow) (hematoxylin and eosin, ×40). (b) Mesonephric adenocarcinoma with endometrioid carcinoma like squamous morules (hematoxylin and eosin, ×100). (c) Hyperplasti c mesonephric remnants with small compact tubules, some of which demonstrate pale eosinophilic intraluminal secreti ons (hematoxylin and eosin, original magnificati on ×100). (d) Mesonephric remnants seen deep within myometrium (thin arrow) adjacent to a calcified myometrial vessel (hematoxylin and eosin, ×40)

Click here to view
Figure 2: (a) Immunohistochemistry in mesonephric adenocarcinoma and mesonephric hyperplasia [indirect immunoperoxidase (IIP)]. Strong and diff use cytokerati n 7 positi vity in mesonephric hyperplasti cremnants (IIP, ×40). (b) and focal cytokerati n 7 in adjacent mesonephric adenocarcinoma (IIP, × 40). (c) The mesonephric remnants and adenocarcinoma (inset) were immunonegati ve with estrogen receptor, whereas cervical stroma showed nuclear positi vity (IIP, ×40). (d) Luminal pattern of CD10 positivity in hyperplastic mesonephric remnants (IIP,×100)

Click here to view


Bilateral adnexal masses were histologically serous cystadenomas and bilateral pelvic lymph nodes were negative for metastasis. The patient received adjuvant radiotherapy (brachytherapy) and is recurrence free 6 months postoperatively.


   Discussion Top


Mesonephric adenocarcinoma is one of the rarest subtypes of cervical adenocarcinoma derived from remnants of the mesonephric ducts. [2],[4] Although it is recognized as a distinct subtype of adenocarcinoma in the World Health Organization's histologic classification of cervical epithelial tumors, very few cases have been reported in literature. [4],[5]

Mesonephric remnants consist of small cysts and/or tubules lined by bland cuboidal or low columnar nonciliated epithelium. The cytoplasm may appear clear or eosinophilic, but does not contain mucin (in contrast to endocervical epithelium) or glycogen. Characteristically, a densely eosinophilic, Periodic-acid Schiff-positive luminal secretion is present. Mesonephric hyperplasia, which is presumed to arise from mesonephric remnants, is an uncommon lesion and is most often an incidental finding. Ferry and Scully have proposed a maximum diameter of 6 mm to differentiate between mesonephric remnants and hyperplasia. [2] Mesonephric hyperplasia has been further divided into three categories: Lobular mesonephric hyperplasia, diffuse mesonephric hyperplasia, and pure mesonephric ductal hyperplasia. [2] The differential diagnosis of mesonephric hyperplasia includes mesonephric carcinoma, endocervical adenocarcinoma, and clear-cell carcinoma. The diffuse form of mesonephric hyperplasia can mimic a well-differentiated adenocarcinoma if it extends deeply into the cervical wall. A back-to-back pattern of glands, malignant nuclear features, and the presence of perineural or vascular invasion support the diagnosis of carcinoma. [3] In the present case, features of adenocarcinoma were present adjacent to hyperplastic remnants and the other unusual feature, hitherto undescribed, was the presence of these mesonephric remnants deep in the myometrium.

The diagnosis of malignant mesonephric tumors is challenging, as the pathologists need to be aware of the various morphologic patterns seen in these group of tumors. When mesonephric duct remnants and/or hyperplasia are found in the vicinity of the malignant neoplasm, as seen in our case, the diagnosis is usually straightforward. In a study of 11 cases of mesonephric adenocarcinoma, histological patterns described are ductal, tubular, retiform, solid, and a sex cord-like pattern. In 10 of these 11 cases, there were hyperplastic mesonephric remnants adjacent to the neoplastic element. [4] In the present case, the presence of squamous morules in the adenocarcinoma prompted us to include a primary endometrioid adenocarcinoma in the differential diagnosis. However, the associated mesonephric hyperplasia coupled with the lack of immunoreactivity to ER and PR (as expected in an endometrioid adenocarcinoma) favored the diagnosis of mesonephric adenocarcinoma.

Immunohistochemical evaluation may aid in differentiating these tumors from their Mullerian counterparts. Lang and Dallenbach-Hellweg [6] suggested that anti-CEA and anti-vimentin immunohistochemical stains would be useful in the differential diagnosis between cervical adenocarcinomas of mesonephric and Mullerian origin, the former being CEA negative and vimentin positive. Luminal CD10 positivity has also been shown in cervical mesonephric remnants and this antibody has been suggested as a useful immunohistochemical marker of mesonephric lesions in the female genital tract. Positive CD10 staining may be useful in confirming a mesonephric origin. Most other benign endocervical glandular lesions were totally negative with CD10. [7],[8] The present case also demonstrated characteristic CD10 luminal pattern of positivity, better appreciated in the hyperplastic mesonephric tubules. In a study of eight cases, most mesonephric adenocarcinomas were immunoreactive for vimentin and calretinin and negative for estrogen and progesterone receptors and CEA. [5] ER and PR were negative in our case.

Recently, PAX2 has been demonstrated in mesonephric remnants and in mesonephric hyperplasia, but not in adenocarcinomas of mesonephric origin. [9] PAX2 was not done in the present case due to non-availability of the antibody at our institute.

The biologic behavior, prognosis, and treatment strategies of this unusual tumor remain uncertain with some mesonephric adenocarcinomas displaying an aggressive clinical course. As there are no definite recommendations of a particular course of therapy for this uncommon disease, it would be reasonable to manage patients with mesonephric adenocarcinoma of the cervix according to current guidelines for cervical adenocarcinoma of similar stage and pathologic findings. [10] Hence, the present patient was managed with postoperative brachytherapy as per the guidelines.

In conclusion, we have reported a case of cervical mesonephric adenocarcinoma of endometrioid morphology with associated hyperplastic mesonephric remnants. A pathologist needs to be aware of this unusual entity with its various patterns so as to arrive at an accurate diagnosis.


   Acknowledgment Top


The authors would like to acknowledge Dr Munita Bal, Associate Professor, Department of Pathology, Tata Memorial Hospital, Mumbai, for being associated with diagnosis of this case.

 
   References Top

1.Hart WR. Symposium part II: Special types of adenocarcinoma of the uterine cervix. Int J Gynecol Pathol 2002;21:327-46.  Back to cited text no. 1
    
2.Ferry JA, Scully RE. Mesonephric remnants, hyperplasia and neoplasia in the uterine cervix. A Study of 49 cases. Am J Surg Pathol 1990;14:1100-11.  Back to cited text no. 2
    
3.Bague S, Rodríguez IM, Prat J. Malignant mesonephric tumors of the female genital tract: A clinicopathologic study of 9 cases. Am J Surg Pathol 2004;28:601-7.  Back to cited text no. 3
    
4.Silver SA, Devouassoux-Shisheboran M, Mezzetti TP, Tavassoli FA. Mesonephric adenocarcinomas of the uterine cervix: A study of 11 cases with immunohistochemical findings. Am J Surg Pathol 2001;25:379-87.  Back to cited text no. 4
    
5.Clement PB, Young RH, Keh P, Ostör AG, Scully RE. Malignant mesonephric neoplasms of the uterine cervix. A report of eight cases, including four with a malignant spindle cell component. Am J Surg Pathol 1995;19:1158-71.  Back to cited text no. 5
    
6.Lang G, Dallenbach-Hellweg G. The histogenetic origin of cervical mesonephric hyperplasia and mesonephric adenocarcinoma of the uterine cervix studied with immunohistochemical methods. Int J Gynecol Pathol 1990;9:145-57.  Back to cited text no. 6
    
7.McCluggage WG, Oliva E, Herrington CS, McBride H, Young RH.CD10 and calretinin staining of endocervical glandular lesions, endocervical stroma and endometrioid adenocarcinomas of the uterine corpus: CD10 positivity is characteristic of, but not specific for, mesonephric lesions and is not specific for endometrial stroma. Histopathology 2003;43:144-50.  Back to cited text no. 7
    
8.Ordi J, Nogales FF, Palacin A, Márquez M, Pahisa J, Vanrell JA, et al. Mesonephric adenocarcinoma of the uterine corpus: CD10 expression as evidence of mesonephric differentiation. Am J Surg Pathol 2001;25:1540-5.  Back to cited text no. 8
    
9.Rabban JT, McAlhany S, Lerwill MF, Grenert JP, Zaloudek CJ . PAX2 distinguishes benign mesonephric and mullerian glandular lesions of the cervix from endocervical adenocarcinoma, including minimal deviation adenocarcinoma. Am J Surg Pathol 2010;34:137-46.  Back to cited text no. 9
    
10.Sedlis A, Bundy BN, Rotman MZ, Lentz SS, Muderspach LI, Zaino RJ.A randomized trial of pelvic radiation therapy versus no further therapy in selected patients with stage IB carcinoma of the cervix after radical hysterectomy and pelvic lymphadenectomy: A Gynecologic Oncology Group Study. Gynecol Oncol 1999;73:177-83.  Back to cited text no. 10
    

Top
Correspondence Address:
Santosh Menon
Department of Pathology, 8th Floor, Annexe Building, Tata Memorial Hospital, Dr. Ernest Borges Marg, Parel, Mumbai, Maharashtra
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.116150

Rights and Permissions


    Figures

  [Figure 1], [Figure 2]

This article has been cited by
1 Mesonephric adenocarcinoma of the cervix: Case report and literature review
A. Dierickx,M. Göker,G. Braems,P. Tummers,R. Van den Broecke
Gynecologic Oncology Reports. 2016; 17: 7
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
   Acknowledgment
    References
    Article Figures

 Article Access Statistics
    Viewed4697    
    Printed106    
    Emailed0    
    PDF Downloaded112    
    Comments [Add]    
    Cited by others 1    

Recommend this journal