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  Table of Contents    
CASE REPORT  
Year : 2014  |  Volume : 57  |  Issue : 4  |  Page : 614-616
Paratesticular papillary serous cystadenocarcinoma: A rare entity in Indian population


1 Department of Pathology, Dayanand Medical College and Hospital, Ludhiana, Punjab, India
2 Department of Urology, Dayanand Medical College and Hospital, Ludhiana, Punjab, India

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Date of Web Publication11-Oct-2014
 

   Abstract 

Ovarian-type surface epithelial carcinomas of testis and paratestis are very rare. These tumors develop from mesothelial inclusions or abnormalities in the development of coelomic epithelium. The diagnosis of these lesions can be difficult, both clinically and radiologically, as morphological, immunohistochemical and ultrastructural features overlap with serous tumors arising in the female genital tract. The pathologist should be aware of the histopathological features and immunostains which help in reaching definite diagnosis. To the best of our knowledge only one such case has been reported in Indian literature. We hereby report a case of young male with this unusual lesion diagnosed on histopathology.

Keywords: Mullerian tumors, paratestis, serous cysteadenocarcinoma, testis

How to cite this article:
Narang V, Sood N, Garg B, Aulakh BS. Paratesticular papillary serous cystadenocarcinoma: A rare entity in Indian population . Indian J Pathol Microbiol 2014;57:614-6

How to cite this URL:
Narang V, Sood N, Garg B, Aulakh BS. Paratesticular papillary serous cystadenocarcinoma: A rare entity in Indian population . Indian J Pathol Microbiol [serial online] 2014 [cited 2019 Dec 13];57:614-6. Available from: http://www.ijpmonline.org/text.asp?2014/57/4/614/142696



   Introduction Top


Ovarian-type surface epithelial carcinomas of testis and paratestis are very rare. [1] Histologically, these are classified as borderline micro-invasive serous tumor and serous carcinoma, intra and paratesticular mucinous tumor with different grades, clear cell and endometrioid adenocarcinoma and transitional cell tumors. The most frequent histologic type is serous. Less than 50 cases of testicular and paratesticular serous type have been reported in the English literature so far. To the best of our knowledge only one such case has been reported in Indian literature. [1],[2] Herein, we report our experience with a 17 year male with paratesticular serous cystadenocarcinoma.


   Case Report Top


A 17-year-old Indian male presented with chief complaint of painful right testicular swelling for 6 months. His past medical history was unremarkable. Radiological investigations raised suspicion of teratoma. Serum AFP (0.97 IU/ml), Beta HCG (0.17 IU/ml) and LDH (153) were within normal limits. High inguinal orchidectomy was planned and specimen was sent for histopathological evaluation.

Gross findings

After orchiectomy, the received specimen in the pathology department showed a paratesticular mass measuring 3 2.5 2 cm with an intact capsule. No necrosis or hemorrhage was noted. Few yellowish nodules were identified in the tumor area [Figure 1]a.
Figure 1: (a) Gross photograph depicting compressed testicular parenchyma and paratesticular tumor (arrow). (b) Paratesticular papillary tumor infiltrating the normal testicular parenchyma (100×, H and E). (c) Cuboidal cells arranges in papillary fashion with cells exhibiting mild nuclear pleomorphism and psammoma body (arrow) (400×,H and E). (d) Tumor cells positive for cytokeratin 7 (CK7)

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Microscopic examination

The tumor sections showed papillary projections lined by atypical cuboidal cells with basophilic cytoplasm. There were many psammoma bodies. The tumor was seen infiltrating the testicular parenchyma. All the histologic findings were typically identical to ovarian papillary serous cystadenocarcinoma. Immunohistochemistry was performed for confirmation and to exclude mesothelioma of tunica vaginalis. The tumor cells were reactive for cytokeratin (CK), cytokeratin 7 (CK7), estrogen receptor but nonreactive with CK20, Vimentin and S100 [Figure 1]b-d.


   Discussion Top


The paratesticular region includes the testicular collecting system, the testicular tunics, and spermatic cord. The rete testis is also considered part of the paratestis although it is principally intratesticular in location. A wide variety of cysts, hyperplasias, neoplasms, and tumor-like conditions may affect the paratesticular region, at times resulting in challenging problems in differential diagnosis. [3]

Mullerian epithelial tumors of testis and paratestis are uncommon, the most common of which are serous type. [4],[5] These serous papillary tumors are benign, borderline, or invasive (malignant). They have morphologic, immunohistochemical and ultrastructural features of serous tumors arising in the female genital tract. [5],[6] These tumors develop from mesothelial inclusions or abnormalities in the development of coelomic epithelium i.e., from areas of coelomic epithelium that became trapped in the testicular tissue. [3],[4],[5] The presenting signs and symptoms in the previously reported cases have been hydrocele, and testicular fullness or mass. [2],[5] Our case presented with testicular fullness with dull pain for several months. Our case showed tumor in paratesticular region with gross and microscopic features exactly similar to ovarian serous cystadenocarcinoma and numerous psammoma bodies. Mitotic figures were inconspicuous and tumor was seen to be infiltrating the testicular parenchyma. Morphologically most important differential diagnosis was mesothelioma arising from tunica vaginalis. [6] More than 60 cases of tunica vaginalis mesothelioma have been reported. The tumor occurs over a broad age range and has features which may overlap with papillary serous carcinoma but is more commonly seen in older individuals. Also, in contrast to the serous carcinoma, history of asbestos exposure is often seen in 40% cases and presenting complaint is hydrocoele. The present case had neither of these. [4],[5],[6]

Immunohistochemical expression of pankeratin, CAM 5.2, CK7, CK903, Ber-EP4, Vimentin, S100, and CEA and virtually no expression of CK5/6, CK20, calretinin, thrombomodulin, or glypican 3 helps in differentiating serous papillary cystadenocarcinoma for malignant mesotheliomas in this region. [7] In the present case tumor cells were reactive for cytokeratin (CK), cytokeratin 7 (CK7), estrogen receptor but nonreactive with CK20, Vimentin and S100.

Microscopically they are distinguished from serous carcinomas by more tubular, less papillary growth pattern; lack of columnar cells and no psammoma bodies. In the present case morphological and immunohistochemical findings were against this diagnosis. Another important issue is the diagnosis of benign, borderline or malignant nature of the papillary serous tumors. [5],[7] In our case overt stromal invasion, made this differential diagnosis easy.

Adenocarcinomas of rete testis and epidiymis are two rare neoplasms which can occur in paratesticular region. According to Nochomovitz, the adenocarcinoma of rete testis should be distinguished from serous carcinomas by more elongated, branching tubules and solid areas, absent or rare psammoma bodies and less frequent papillae. The literature also support the idea that metastatic carcinomas must be excluded before a primary adenocarcinoma of epididymis is suspected. The primary carcinomas of epididymis have cells which are clear and glycogenated and psammoma bodies are absent. [8],[9]

Reported mean age of serous papillary carcinoma is 56 (14-77) for borderline and 31 (16-42) years for invasive forms. [6] The present case was 17-years-old young male. The recommended treatment is radical orchidectomy, because the tumor is highly resistant to radiotherapy and chemotherapy. [8],[9],[10] Our case was successfully treated with radical orchiectomy and is on follow-up. Long-term follow up of the patients with this tumor is important because there is high probability of long-term metastasis. [2]

In conclusion, serous cystadenocarcinomas of male genital tract are very similar to their ovarian counterpart but their occurrence in the male genital tract is extremely rare. However, it should be considered in the differential diagnosis of any malignant testicular tumor with epithelial pictures.



 
   References Top

1.
Kurian RR, Prema NS, Belthazar AA. Paratesticular papillary serous cystadenocarcinoma: A case report. Indian J Pathol Microbiol 2006;49:36-7.  Back to cited text no. 1
[PUBMED]    
2.
Maruschkem M, Schmidt W, Casper J, Hakenberg OW. Ovarian type surface epithelial carcinoma of the testis with delayed metastatic spread. Urol Int 2008;81:119-21.  Back to cited text no. 2
    
3.
Srigley JR. The paratesticular region: Histoanatomic and general considerations. Semin Diagn Pathol 2000;17:258-69.  Back to cited text no. 3
[PUBMED]    
4.
Leonardo C, Franco G, Michetti M, De Nunzio C, Zampelli A, De Dominicis C. A rare case of serous papillary cystadenocarcinoma of testis. J Androl 2010;31:434-6.   Back to cited text no. 4
    
5.
Amin MB. Selected other problematic testicular and paratesticular lesions: Rete testis neoplasms and pseudotumos, mesothelial lesions and secondary tumors. Mod Pathol 2005;18:S131-45.  Back to cited text no. 5
[PUBMED]    
6.
Geramizadeh B, Farzaneh MR, Pakbaz S, Zeighami S. Testicular papillary serous cystadenocarcinoma: A rare case report. Rare Tumors 2011;3:e44. [Published online 2011 Dec 23].  Back to cited text no. 6
    
7.
Anchala PR, Dhir R, Parwani AV, Zynger DL. Immunohistochemical profile of paratesticular serous papillary adenocarcinoma and tunica vaginalis facilitates distinction from malignant mesothelioma. Int J Surg Pathol 2011;19:692-8.   Back to cited text no. 7
    
8.
Sumrall A, Puncky L, Brown A, Thigpen T. Ovarian cancer in a man? Clin Ovarian Cancer 2009;2:57-9.  Back to cited text no. 8
    
9.
Nochomovitz LE, Orenstein JM. Adenocarcinoma of the rete testis. Review and regrouping of reported cases and a consideration of miscellaneous entities. J Urogenital Pathol 1991;1:11-40.  Back to cited text no. 9
    
10.
Jones MA, Young RH, Srigley JR, Scully RE. Parartesticular serous papillary carcinoma, A report of six cases. Am J Surg Pathol 1995;12:1359-65.  Back to cited text no. 10
    

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Correspondence Address:
Vikram Narang
Department of Pathology, Dayanand Medical College and Hospital, Ludhiana, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.142696

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