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Case Report *Corresponding author

Khoi Anh Nguyen, Department of , Pham Ngoc Thach University of Medicine, Vietnam, Email: Malignant Transformation in a [email protected] Submitted: 16 April 2018 Parotid Warthin’s Tumor: Clinical Accepted: 01 May 2018 Published: 04 May 2018 Features and Histopathological Copyrighta © 2018 Nguyen et al. Examination OPEN ACCESS Keywords Khoi Anh Nguyen1*, Tu Anh Thai2, and Cuong Tri Giang2 • Squamous cell 1Department of Oncology, Pham Ngoc Thach University of Medicine, Vietnam • 2Ho Chi Minh City Oncology Hospital, Vietnam • Warthin’s tumor • Parotid

Abstract Warthin’s tumor is a tumor consisting of epithelial and lymphoid components. The majority of Warthin’s tumors are benign and malignant transformation is extremely rare. We report a case of malignant transformation of a Warthin’s tumor of the right . The patient had a right parotid tumor and underwent tumor excision. Histopathology after surgery showed malignant transformation of Warthin’s tumor. On examination, we found a cervical lymph node group VB and Fine Needle Aspiration (FNA) showed metastasis. We decided to perform a total and modified radical right neck dissection for the patient. Then the patient underwent adjuvant radiotherapy. Close follow-up was carried out and 3 months after surgery there was no evidence of recurrence or metastatic . Therefore, if there is a parotid tumor which is suspected malignant, we should thoroughly investigate the cervical lymph nodes and consider prophylactic neck dissection although we do not find the susceptive nodes.

BACKGROUND CASE REPORT

Warthin’s tumor (adenolymphoma) was first described by A 63-year-old man had a painless mass of about 0.5cm in size Aldred Warthin in 1929. Warthin’s tumor accounts for about located in the right auricular region. It had been present for the 5-10% of all parotid tumors and it is the second most common past year, but grew rapidly during the last two months. In those benign tumor of the salivary . Warthin’s tumor is more two months, the mass grew to approximately 7cm. Fine Needle common in men with the average age range from 60 to 70 years. It Aspiration (FNA) was performed and the smears showed the is also related to smoking. The tumor normally has no symptoms epithelial cells with atypical nuclei. The patient then underwent and begins as a slow growing nodular, indolent mass that is firm tumor excision. Histopathology after surgery showed malignant or fluctuant at palpation and about10% of the cases appear on transformation of Warthin’s tumor, the malignancy showing both sides [1-5]. adenosquamous and squamous carcinoma features, margins negative. A sudden increase in tumor size may be associated with inflammation or malignant transformation [1,2]. The malignant The patient was transferred to Ho Chi Minh City Oncology transformation of Warthin’s tumor is extremely rare and occurs Hospital. At the examination, we found only the right auricular in about 0.3% of cases. The transformation of the lymphoid region with mild edema, paralysis of the marginal mandibular component to a malignant lymphoma appears to occur more branch and other organs not unusual. frequently than an epithelial malignant transformation, which is The Doppler ultrasonography showed the right parotid was extremely rare [3,4]. Pathogenesis of malignant transformation partially removed. The inferior pole in the deep partial of the of Warthin’s tumor is unknown [6]. Until 2008, only 32 cases of superficial lobe had two mass about 9mm in size. The cervical epidermoid carcinoma have been reported to arise in Warthin’s B et al. lymph node group V was hypoechoic about 10mm in size, tumor [3].There were 4 cases of as a angiogenesis in the navel and margin. FNA resulted in lymph malignant component have been listed by Therkildsen node metastatic carcinoma (Figure 1). [7], P J Yaranal and Umashankar reported and a case of squamous cell carcinoma which arose in a Warthin’s tumour of the right On histo pathological examination of parotid gland tumor, parotid gland in 2013 [8]. Fabiana Allevi Federico Biglioli the tumor shows one part of characteristic Warthin tumor, reported a case of squamous cell carcinoma arising in Warthin’s which is composed of bilayered oncocytic and tumour in 2014 [9]. The epithelial component evolving into dense lymphocytes background (Figure 2). The continuous , mucoepidermoid carcinoma, squamous cell area of malignant change was observed (Figure 3). The other carcinoma, oncocytic carcinoma and Merkell cell carcinoma have part of malignancy exhibits malignant squamous cells and been documented [6,10-16]. adenosquamous feature with small duct lumens (Figure 4). Cite this article: Nguyen KA, Thai TA, Giang CT (2018) Malignant Transformation in a Parotid Warthin’s Tumor: Clinical Features and Histopathological Examination. J Cancer Biol Res 6(1): 1115. Nguyen et al. (2018) Email: [email protected]

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Figure 1

The image of Doppler ultrasonography.

Figure 2

A: Warthin tunour, which is composed of bilayered oncocytic epithelium lines numerous fluids-containing cystic spaces and lymphoid tissue. H&E, X40 B: Higher magnification of bilayered oncocytic epithelium (arrow) and dense lymphocytes background. H&E, X100.

Figure 3

A: Warthin tumor has characteristic epithelial and lymphoid component (at the left of figure) but, in addition, the continuous area of malignant change was observed (at the right of figure). H&E, X40 B: Immunohistochemistry stained by Cytokeratin, the epithelial component including benign and malignant change is highlight on the lymphocytes background (at the left of figure) and stromal invasion (at the right of figure). CK, X40. DISCUSSION Atypical mitoses are shown in figures (Figure 4).

We performed a total parotidectomy and modified radical The diagnosis of the malignant transformation of Warthin’s right neck dissection for the patient. After surgery, the patient tumor1. to carcinoma is based on the following criteria [8-10]: had paresis of the right facial nerve (Figure 5). Postoperative 2. histology showed squamous cell carcinoma/ adenocarcinoma Presence of a pre-existing benign Warthin’s tumor; grad 2, malignant transformation of Warthin’s tumor and one Presence of transitional zones from benign oncocytic to cervical lymph node (group VB) metastatic with squamous malignant epithelium; cell carcinoma and adenocarcinoma features. The patient subsequently underwent adjuvant radiotherapy with Intensity 3. Presence of an infiltrating growth in the surrounding Modulated Radiation Therapy technique. The doses were 60 Gy lymphoid tissue; in 2 Gy fractions. Close follow-up was carried out and 6 months 4. Exclusion of metastasis to lymphoid stroma from an after surgery there was no evidence of local recurrence or extra-salivary primary carcinoma. metastatic neoplasm (Figure 5). J Cancer Biol Res 6(1): 1115 (2018) 2/4 Nguyen et al. (2018) Email: [email protected]

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Figure 4

Higher magnification of malignancy. A: Higher magnification of malignancy exhibits malignant squamous cells (asterisks) and adenosquamous feature with small duct lumens (arrows). H&E, X200 B: Atypical mitoses (arrows). H&E, X200

Figure 5

Patient after surgery.

Carcinomas arising in Warthin’s tumor are rare. Nagao et nodes when the patient had a malignant Warthin’s tumor and al studied two cases of mucoepidermoid carcinoma arising in considerCONCLUSION prophylactic neck. Warthin’s tumor of the parotid gland [6]. Gunduz et al., reported cases of squamous cell carcinoma arising in Warthin’s tumor [7]. Fornelli et al., reported two cases of Merkell cell carcinoma of The malignant transformation of Warthin’s tumor is rare. the parotid gland associated with Warthin’s tumor [12]. Seifert Diagnosis is based on histopathology and immunohistochemistry. described bilateral mucoepidermoid arising in In clinical observations, we should be suspicious of patients with previous Warthin’s tumor, recent fast-growing tumors, and bilateral pre-existing Warthin’s tumor of the parotid gland [2]. REFERENCES In reported cases, one third showed metastasized regional lymph evidence of cervical lymph node metastasis with FNA. nodes and one case metastasized by blood to the lung and liver 1. [9,14,17]. Auclair PL, Ellis GL, Gnepp DR. Other benign epithelial . Clinically, our patient had a mass which enlarged rapidly Surgical Pathology of the Salivary Gland. In: Ellis GL, Auclair PL, Gnepp 2. within two months. The tumor size is 14 times greater than the DR. Philadelphia, WB Saunders Company. 1991; 252-268. original (7cm vs 0.5cm). This is the major factor to consider for Siefert G. Carcinoma in pre-existing Warthin’s tumors malignancy. (cystadenolymphoma) of the parotid gland. Classification, pathogenesis and differential diagnosis. Pathologe. 1997; 18: 359-367. Malignant diagnosis requires evidence of stromal invasion, 3. Sharama M, Chintamani, Saxena S, Agrawal S. Squamous cell local invasion or lymph node metastasis [6,9]. Our patient was carcinoma arising in unilateral Warthin’s tumour of the parotid gland. diagnosed by local invasion on histopathology and cervical J Oral Maxillofacial Pathol. 2008; 12: 82-84. lymph node metastasis by FNA. Therefore, we suggest that the physicians should thoroughly investigate the cervical lymph 4. Cob CJ, Greaves TS, Raza AS. Fine needle aspiration cytology J Cancer Biol Res 6(1): 1115 (2018) 3/4 Nguyen et al. (2018) Email: [email protected]

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Cite this article Nguyen KA, Thai TA, Giang CT (2018) Malignant Transformation in a Parotid Warthin’s Tumor: Clinical Features and Histopathological Examination. J Cancer Biol Res 6(1): 1115.

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