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CASE REPORT  
Year : 2011  |  Volume : 54  |  Issue : 3  |  Page : 574-577
High-grade adenocarcinoma, (ductal type) arising in unilateral Warthin tumor of the parotid gland


1 Department of Pathology, Tata Memorial Hospital, Parel, Mumbai, India
2 Junior Histopathologist, SRL Diagnostics, Tata Memorial Hospital, Parel, Mumbai, India
3 Surgical Oncology, Tata Memorial Hospital, Parel, Mumbai, India

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Date of Web Publication20-Sep-2011
 

   Abstract 

Warthin tumor is a well-recognized benign salivary gland neoplasm consisting of an epithelial as well as a lymphoid component. Malignant transformation in Warthin tumor is rare and its reported incidence is up to 1%. The more common types of carcinomas described in Warthin tumor are the squamous and mucoepidermoid types, with high-grade adenocarcinoma being extremely rare. A high-grade adenocarcinoma (ductal type) arising in the Warthin tumor in a 72-year-old man is presented for its rarity and diagnostic difficulties.

Keywords: High-grade adenocarcinoma, parotid gland, Warthin tumor

How to cite this article:
Deodhar KK, Shah M, Chaturvedi P. High-grade adenocarcinoma, (ductal type) arising in unilateral Warthin tumor of the parotid gland. Indian J Pathol Microbiol 2011;54:574-7

How to cite this URL:
Deodhar KK, Shah M, Chaturvedi P. High-grade adenocarcinoma, (ductal type) arising in unilateral Warthin tumor of the parotid gland. Indian J Pathol Microbiol [serial online] 2011 [cited 2020 Dec 2];54:574-7. Available from: https://www.ijpmonline.org/text.asp?2011/54/3/574/85097



   Introduction Top


Warthin tumor ( syn. Adenolymphoma, papillary cystadenoma lymphomatosum,) is the second-most common benign salivary gland tumor and characteristically presents in the lateral lobe of the parotid gland. It accounts for approximately 5 to 15% of all parotid tumors. [1] The tumor may develop at any age, although it is rare under the age of 40, and shows slight male preponderance. [1],[2] Malignant transformation of the Warthin tumor is very rare. The incidence of carcinoma ex Warthin tumor is up to 1%. [1],[3] Among the carcinomas seen in Warthin tumor, the more common ones are the squamous [4],[5] and mucoepidermoid types. [6],.[7],[8] High-grade adenocarcinoma has been very seldom described. [9],[10],[11] Extreme rarity and challenges faced during diagnosis prompted us to report this case.


   Case Report Top


A 75-year-old man presented with a swelling in the right cervical level II region, since the last two years. Fine needle aspiration cytology (FNAC) was done outside and reported as metastatic squamous carcinoma. This diagnosis, on review, was retained in our hospital. Clinical and radiological examination, including a Computerized Tomography (CT) scan of the head and neck/para nasal sinuses, and X-ray chest, did not reveal any obvious primary site. A CT scan of the head and neck region revealed an enhancing oval soft tissue mass measuring 2.3 × 1.4 cm, most likely representing an enlarged right level II lymph node. The possibility of this lesion being the deep lobe of the parotid was also kept in mind. The superficial lobes of both parotids were normal.

A repeat Ultrasonography (USG) guided FNAC was performed on this? enlarged level II lymph node/? deep lower pole of the parotid gland; and a diagnosis of Warthin tumor was made. On account of the discrepancy between the two FNAC reports and a non-conclusive CT scan report, surgery with frozen section diagnosis was planned. A total parotidectomy with right level II lymph node sampling was performed.

Gross examination showed a well-encapsulated tumor measuring 3 × 3 × 2 cm, with a normal parotid gland measuring 1.2 × 1.2 × 1 cm. The cut surface was solid with tiny sub-centimeter-sized cystic spaces. On frozen section, a diagnosis of Warthin tumor was made and the level II neck lymph nodes were found to be reactive. As the initial FNAC was reported as squamous carcinoma, and in view of the frozen section report, the entire tissue was processed for a final histology.

Histology showed the majority of the tumor to be composed of glandular, cystic, and often papillary structures, lined by oncocytic epithelial cells and lymphoid stroma, with abundant follicles - typical of the Warthin tumor [Figure 1]a to d. However, in addition, a focus of carcinoma (maximum diameter 13 mm) was seen, showing infiltration of the stroma and focally showing a vague glandular pattern. The tumor cells had large vesicular nuclei and eosinophilic cytoplasm [Figure 3],[Figure 4]a and b. This focus was seen in only one out of six slides. A transition between the benign and malignant epithelium was also identified, in the form of cellular atypia/disarray in the Warthin tumor [Figure 2]. On immunohistochemistry, the tumor cells expressed AE1/AE3, CK7 [Figure 4]d, and weakly Ber EP4 [Figure 4]c. They were negative for CK20. All the eight right level II nodes were negative for metastasis. Considering that the tumor showed a vague glandular pattern, coupled with CK7 and BerEP4 positivity on Immunohistochemistry (IHC); a diagnosis of high-grade ductal type adenocarcinoma arising in Warthin tumor was made.
Figure 1: (a) Warthin tumor with adjacent normal salivary gland (H and E, x25), (b) Warthin tumor with adjacent normal salivary gland (H and E, x25), (c) Warthin tumor (H and E, x100), (d) Warthin tumor (H and E, x100)

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Figure 2: Cytological atypia/cellular disarray in Warthin tumor

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Figure 3: High-grade ductal adenocarcinoma area (H and E, x100)

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Figure 4: (a) Malignant epithelium and adjacent benign ducts (H and E, x215;100), (b) High-grade ductal adenocarcinoma area (H and E, x200), (c) IHC BerEP4 x200, (d) IHC CK7 x200

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Follow up : T0 he patient was advised radiotherapy, which he declined to take. Currently, the patient is well, 16 months after the surgery. No recurrence or metastasis is noted.


   Discussion Top


Malignant change in Warthin tumor is very rare.

Epithelial malignancy in aWarthin tumor can be seen in three forms : F0 irst, the tumor arising within the Warthin tumor; second, a metastatic carcinoma seen in an intraparotid node; and third, a coexistent neoplasm along with the Warthin tumor. The types of carcinomas seen in the Warthin tumor are squamous carcinoma [4],[5] and mucoepidermoid carcinoma. [6],[7],[8] Adenocarcinomas are extremely rarely described [9],[10],[11] ; in that tumors with oncocytic features are identified. [9],[10] Of late, a new tumor - 'Warthin adenocarcinoma' with a striated duct phenotype was described. [11]

A change in phenotype of cells through to squamous metaplasia and goblet cell metaplasia is a well-known feature of the Warthin tumor [7] and may thus explain the occurrence of epidermoid carcinoma and mucoepidermoid carcinoma. However, squamous metaplasia, per se, should not be mistaken as a malignant change or metastasis. The occurrence of adenocarcinomas in the Warthin tumor is also plausible as oncocytic tumors show glandular differentiation. [10]

Our case showed histology of the adenocarcinoma. Due to the rarity of carcinoma in the Warthin tumor, a metastasis from another site had to be excluded. There was no obvious primary in the head and neck region on clinical examination and CT scan. The tumor cells expressed AE1/AE3, CK7 and BerEP4; while they were negative for CK20. The latter ruled out a lower gastrointestinal tract (GIT) primary. Adenocarcinomas from the upper GIT and the pancreaticobiliary tree usually expressed both CK7 and CK 20 positivity, hence, a primary from these sites was reasonably excluded.

Histologically, a transition from benign to malignant glands was well seen, in the form of cellular atypia and disarray in Warthin epithelium. Based on gross and microscopy, a diagnosis of high grade ductal type adenocarcinoma, arising in Warthin tumor is made. The margins could not be accurately commented upon due to tissue fragmentation artifacts.

Review of the initial outside FNAC confirmed a high-grade carcinoma and, in retrospect, was consistent with high-grade ductal adenocarcinoma rather than squamous carcinoma. No keratin pearls were seen.

In conclusion, ductal type adenocarcinoma in Warthin tumor of the parotid gland is a very rare event, but one should remain open for its possibility. Histological overlap between ductal type adenocarcinoma and squamous carcinoma is also to be kept in mind.

 
   References Top

1.Simpson RH, Eveson JW. Warthin tumor. In : B0 arnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and Genetics Head and Neck tumours. World Health Organisation Classification of Tumours. Lyon : I0 ARC Press; 2005. p. 263-5.  Back to cited text no. 1
    
2.Eveson JW, Cawson RA. Salivary gland tumors. A review of 2410 cases with particular reference to histological types, site, age and sex distribution. J Pathol 1985;146:51-8.  Back to cited text no. 2
[PUBMED]    
3.Seifert G, Bull HG, Donath K. Histologic subclassification of the cystadenolymphoma of the parotid gland. Analysis of 275 cases. Virchows Arch A Pathol Anat Histol 1980;388:13-38.  Back to cited text no. 3
[PUBMED]    
4.Skalova A, Michal M, Nathansky Z. Epidermoid carcinoma arising in Warthin's tumour, a case study. J Oral Pathol Med 1994;23:330-3.  Back to cited text no. 4
    
5.Gunduz M, Yamanaka N, Hotomi M, Kuki K, Yokoyama M, Nakamine H. Squamous cell carcinoma arising in a Warthin'stumor. Auris Nasus Larynx 1999;26:355-60.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Nagao T, Sugano I, Ishida Y, Tajima Y, Furuya N, Kondo Y, et al. Mucoepidermoid carcinoma arising in Warthin'stumour of the parotid gland : R0 eport of two cases with histopathological, ultrastructural and immunohistochemical studies. Histopathology 1998;33:379-86.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Yamada S, Matsuo T, Fujita S, Suyama K, Yamaguchi A, Mizuno A. Mucoepidermoid carcinoma arising in Warthin's tumor of the parotid gland. Pathology Int 2002;52:653-6.  Back to cited text no. 7
    
8.Mardi K, Sharma J. Mucoepidermoid carcinoma arising in Warthin's tumor : A0 case report. Indian J Pathol Microbiol 2007;50:331-3.  Back to cited text no. 8
[PUBMED]    
9.Bengoechea O, Sanchez F, Larrinaga B, Martinez-Penuela JM. Oncocytic adenocarcinoma arising in Warthin's tumor. Pathol Res Pract 1989;185:907-11.  Back to cited text no. 9
    
10.Therkildsen MH, Christensen N, Andersen LJ, Larsen S, Katholm M. Malignant Warthin's tumour, a case study. Histopathology 1992;21:167- 71.  Back to cited text no. 10
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11.Bell D, Luna MA. Warthin Adenocarcinoma : A0 nalysis of 2 cases of a distinct salivary neoplasm Ann Diagn Pathol 2009;13:201-7.  Back to cited text no. 11
    

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Correspondence Address:
Kedar K Deodhar
Department of Pathology, Tata Memorial Hospital, Dr. E. Borges Road, Parel, Mumbai - 400 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.85097

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    Figures

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

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