Synchronous oncocytoma and Warthin’s tumor in the ipsilateral parotid gland

Synchronous oncocytoma and Warthin’s tumor in the ipsilateral parotid gland

Auris Nasus Larynx 31 (2004) 73–78 Case report Synchronous oncocytoma and Warthin’s tumor in the ipsilateral parotid gland Yasutomo Araki a,∗ , Ryoh...

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Auris Nasus Larynx 31 (2004) 73–78

Case report

Synchronous oncocytoma and Warthin’s tumor in the ipsilateral parotid gland Yasutomo Araki a,∗ , Ryohei Sakaguchi b a

Department of Otorhinolaryngology, Keio University School of Medicine, 35 Shinanomachi Shinjuku-ku, Tokyo 160-8582, Japan b Sakaguchj Clinic of Otorhinolaryngology, Kanagawa, Japan Received 29 May 2003; accepted 18 July 2003

Abstract We encountered an 81-year-old Japanese man with oncocytoma of the parotid gland occurring simultaneously with a Warthin’s tumor in the ipsilateral parotid gland. The patient complained of swelling in the right infra-auricular region. Computed tomographic images, magnetic resonance images, and ultrasonography showed two masses in the right parotid gland. Technetium 99 m pertechnetate scintigraphy revealed a high accumulation in both tumors. We performed a superficial lobectomy of the parotid gland. Preoperative diagnosis was multiple Warthin’s tumor, but upon histopathological examination Warthin’s tumor proved to coexist with oncocytoma. In general, the presence of synchronous, major, salivary gland tumors with different histopathology in the ipsilateral parotid gland appears to be unique. This is especially true for the combination of oncocytomas and Warthin’s tumors, which is extremely rare. Only eight cases of this kind coincidence were found in our review of the literature. The similarity of oncocytomas and Warthin’s tumors based on epidemiology, image diagnostics, and histopathology is discussed. © 2003 Elsevier Ireland Ltd. All rights reserved. Keywords: Oncocytoma; Adenolymphoma; Multiple tumors; Salivary gland

1. Introduction

2. Case report

The occurrence of parotid gland tumors are relatively frequent among reported head and neck lesions. Usually, these tumors are solitary, although multiple-occurring tumors are occasionally observed. Most multiple parotid tumors are histologically classified as Warthin’s tumors, some are classified as multiple metastatic parotid tumors and others benign tumors that become multiple malignant tumors [1,2]. The simultaneous occurrence of multiple tumors in the same parotid gland is rare [3]. The coexistence of Warthin’s tumors and oncocytomas is extremely rare, only eight cases were found in our literature review. Here, we describe an interesting case of oncocytoma of the parotid gland occurring simultaneously with a Warthin’s tumor. The clinicopathological relationship between these tumors is also discussed.

An 81-year-old Japanese man presented with a persistent swelling in the right infra-auricular region without pain, experienced this swelling for two months prior to his visit to our office. Physical examination revealed a 2.0 cm × 2.5 cm, elastic, soft, mobile mass with a smooth surface in the right parotid gland. There were no palpable cervical lymph nodes and no facial nerve paralysis. Two years before consultation, he had undergone superficial lobectomy of the left parotid gland for Warthin’s tumor. Ultrasonography revealed two apparent tumors in the right parotid gland, the echo pattern of these tumors indicated that they were benign. Both tumors measured 1.5 cm × 1.5 cm. Again, no palpable cervical lymph nodes were found. Enhanced computed tomographic (CT) images revealed two distinct tumors, one in the lower and the other in the middle portion of the parotid gland, both were round and slightly enhanced (Fig. 1). CT revealed no qualitative differences between the tumors. These tumors produced weak signals in T1- and T2-weighted magnetic resonance imaging (MRI) compared to those produced by the parotid

∗ Corresponding author. Tel.: +81-3-3353-3003; fax: +81-3-3353-3003. E-mail address: [email protected] (Y. Araki).

0385-8146/$ – see front matter © 2003 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.anl.2003.07.008

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Fig. 1. Enhanced contrast CT image (axial) at the level of the parotid gland. Two separate, round, sharply defined, and homogeneously enhanced masses are clearly visible in the right parotid gland.

gland itself (Fig. 2a and b). Both tumors were slightly and homogeneously enhanced in gadolinium-enhanced MRIs. Similar to CT, MRI revealed no tumor differences. Galium 67 m scintigraphy showed no abnormal uptake, but technetium 99 m pertechnetate scintigraphy showed strong accumulation in the lower portion of the parotid gland (Fig. 3). These findings suggested that both tumors were benign. The patient was provisionally diagnosed with multiple Warthin’s tumor in the ipsilateral parotid gland. In September 2000, the patient underwent superficial lobectomy of the parotid gland under our care, his facial nerve was preserved. The resected masses were solid and reddish in gross appearance. Macroscopically there was no difference between the two tumors. Microscopic examination revealed, however, that the two tumors were different histopathologically. Detailed examination of multiple histological sections revealed that one tumor consisted of cystic spaces lined with a double layer of cuboidal to tall columnar eosinophilic epithelial cells. The stroma of this neoplasm contained well-defined lymphoid aggregates (Fig. 4). These findings were consistent with Warthin’s tumor. The other tumor consisted of epithelial cells with abundant granular, eosinophilic cytoplasm and these had uniform round-to-oval nuclei. Lymphoid infiltration of the parenchyma was absent, and intra-parotid lymph node involvement was also absent (Fig. 5a). In addition, phosphotungstic acid–hematoxylin (PTAH) staining showed deep-blue cytoplasmic granules in the epithelial cells. Ultrastructurally, abundant mitochondria were seen in the cytoplasm. They were tightly

packed together and occupied nearly all the cytoplasmic area (Fig. 5b). This histopathologic pattern is identical to that of oncocytomas. Subsequent follow-up for about 2 years revealed no evidence of a recurrent neoplasm.

3. Discussion The histopathological difference between oncocytomas and Warthin’s tumors is the presence or absence of lymphoid stroma. Although these tumors are readily distinguishable via light microscopy [4], they are difficult to differentiate using conventional imaging techniques. Oncocytomas and Warthin’s tumors have very similar imaging features; thus, they are indistinguishable in standard CT and MR images [5]. For example, unlike with other salivary gland tumors, both oncocytomas and Warthin’s tumors exhibit intense accumulation and prolonged retention of technetium 99 m pertechnetate [6]. Thus, limitations of conventional imaging techniques must be considered carefully when used to diagnose parotid gland tumors. Cytological analyses of parotid gland aspirates often provide important information for tumor diagnosis. However, even when tumor specimens are collected with a fine needle, oncocytomas and Warthin’s tumors cannot be distinguished, because the epithelial component of these tumors is almost the same. Even so, the sensitivity of cytological diagnosis for oncocytomas is 29% [7], whereas that for all salivary gland neoplasms is 82–92% [8,9]. Diagnostic errors

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Fig. 2. Magnetic resonance images at the level (axial) of the parotid gland. Axial contrast T1- (a) and T2-weighted (b) images show two less-intense masses in right parotid gland, in agreement with CT scan.

sometimes occur because of the rarity of oncocytomas, limited breadth of interpreter experience, and variable absence of typical features of Warthin’s tumors, such as lymphoid stroma [10]. The incidence of multiple salivary gland tumors is very rare, accounting for ∼1.3% of all salivary [1] and ∼0.4% of all parotid gland tumors [2]. Warthin’s tumors comprise the majority of multiple-occurring tumors [3], 12% of patients

that are present with a single Warthin’s tumor will develop more than one unilateral or bilateral lesion metachronously or synchronously [11]. Coexistence of ipsilateral salivary gland tumors of different histologic type, however, is uncommon, such tumors comprise less than 0.3% of all salivary gland neoplasms [3]. Coexistence of oncocytoma and Warthin’s tumor is extremely rare. Only eight subjects presenting both of these tumors were found in our review

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Fig. 3. Technetium 99 m pertechnetate scintigraphy image. Static scintigraph of the salivary glands show intensely elevated uptake of technetium 99 m pertechnetate in the right parotid gland.

of the English literature [3,7,15–18]. The tumors of all of these subjects localized to the parotid gland, the great majority occurred in the right side. All of these subjects were elderly men (mean age: 65.5 years), and all underwent parotidectomy. Parotid tumors did not recur in any of these subjects. Many authors have pointed out the similarity of oncocytomas and Warthin’s tumors [4,11]. From an etiologic view, these tumors occur most frequently in the sixth and seventh decade [11,12], while other tumors often occur at younger ages [13]. The multicentricity of these tumors has been recognized—7% of oncocytomas [12] and 12% of Warthin’s tumors are multicentric [11]. Both oncocytomas and Warthin’s tumors accumulate technetium 99 m pertechnetate [6]. In addition, both of these tumors show gradual clinical manifestations, follow favorable courses, and are cured by surgical resection [9,11]. Histopathologically, epithelial cells of oncocytomas and Warthin’s tumors contain a large number of mitochondria in the cytoplasm [9,11]. Intermediate types of Warthin’s tumors and oncocytomas have also been reported, in which both tumors share some of the features of the other type. Oncocytomas occasionally contain a few microcysts or macrocysts that have lymphoid infiltrates resembling Warthin’s tumor [14], and Warthin’s tumors occasionally contain foci of nodular oncocytic hyperplasia [4]. This suggests the provisional conclusion that there might be some relationship between these two types of tumors. In the case we encountered and described herein, oncocytoma occurred synchronously with multiple Warthin’s tumors. This brief discussion highlights the common histogenesis of oncocytomas and Warthin’s tumors.

Fig. 4. Histological features of the Warthin’s tumor. The Warthin’s tumor of this patient consisted of multiple cysts lined with a double layer of cuboidal to tall columnar eosinophilic epithelial cells. The stroma contained well-defined lymphoid aggregates. (Hematoxylin–eosin stain; original magnification: 20×).

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Fig. 5. (a) Histological features of the oncocytoma. The oncocytoma of this patient consisted of epithelial cells with abundant granular eosinophilic cytoplasm; the cells’ nuclei were uniform and round-to-oval shaped. No lymphoid infiltration was evident in the parenchyma. (Hematoxylin–eosin stain; original magnification: 100×), (b) ultrastructural features of the oncocytoma. Abundant, tightly packed mitochondria are evident, occupying nearly the entire cytoplasm (original magnification: 5000×).

4. Conclusion We encountered a case of synchronous oncocytoma and Warthin’s tumor in the ipsilateral parotid gland. Simultaneous occurrence of these tumors is extremely rare, only eight cases are previously described in the English literature. Preoperative diagnosis on the basis of conventional imaging was difficult to confirm. The present case report underscores the similarities between oncocytomas and Warthin’s tumors. References [1] Turnbull AD, Frizell EL. Multiple tumors of the major salivary glands. Am J Surg 1969;118:787–9.

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