Uncommon Coexistence of Pleomorphic Adenoma and Warthin's Tumor in a Painfully Swollen Left Parotid Gland: A Surgical Case Report

Abstract

BACKGROUND Benign pleomorphic adenoma is the most common primary tumor of the salivary glands and mainly arises in the parotid gland. Warthin's tumor, or papillary cystadenoma lymphomatosum, represents <30% of benign parotid tumors. The simultaneous occurrence of multiple parotid tumors is rarely described - depending on the corresponding histology (different/identical), the time of their occurrence (synchronous/metachronous), as well as their location (unilateral/bilateral), multiple parotid tumors can be further sub-classified. CASE REPORT We describe the case of a 54-year-old female patient with progressive and painful swelling of the left parotid gland for the last 6 months. During extra-oral examination, a bulging, displaceable mass of approximately 3 cm was determined. A subsequent MRI (magnetic resonance imaging) examination revealed a multifocal lesion but failed to provide a decisive clue as to the tumor entity of the lesion, and a lateral (superficial) parotidectomy was performed. Postoperative histomorphological interpretation allowed the final pathological diagnosis of synchronous, unilateral occurrence of a pleomorphic adenoma as well as a Warthin's tumor. CONCLUSIONS This report presents a rare case of synchronous unilateral parotid tumors and supports that benign pleomorphic adenoma and Warthin's tumor are the most common associations. Since clinical examination, MRI imaging, and even cytological assessment could be misleading in the detection of synchronous ipsilateral multiple parotid gland tumors, our report also highlights the importance of timely and accurate diagnosis with histopathology to plan surgery and to exclude malignant transformation, which is a rare but important association with both types of primary salivary gland tumor.

Figure 1.

Presurgical imaging: Coronal image T1-weighted, fat-saturated of a 54-year-old patient with a smaller contrast-enhancing lesion in the middle part of the parotid gland (histologically proven as a PA, marked by an arrow) and a second less-enhancing lesion in the parotid gland (histologically proven as a WT, marked by an asterisk). PA – pleomorphic adenoma; WT – Warthin’s tumor.

Figure 2.

Presurgical imaging: An ultrasonographical examination revealed a smoothly circumscribed, homogeneous mass (1.4×1.2 cm, marked by an arrow).

Figure 3.

Postoperative histopathology: (A) Adjacent to the lipomatous, berry-shaped glandular fragments of the parotid gland, an encapsulated tumorous lesion was found. A lymphoid stroma with formed germinal centers and an epithelial tumor component are shown. (B) The tumor shows a (mostly) bilayered epithelium – partly cystic, partly solid – attached to a lymphoid stroma. In the upper center of the image, a germinal center is displayed (H&E – hematoxylin and eosin staining).

Figure 4.

Postoperative histopathology: (A) Overview of the preauricular tumor showing a distinct capsule surrounded by acinar glands. (B) The lesion consists of predominant myoepithelial cells (right) adjacent to a myxoid stromal component (left) (H&E – hematoxylin and eosin staining).