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Oncocytic carcinoma of the parotid gland: report of a new case.

Abstract

Prior to 2003, only 39 cases of oncocytic carcinoma of salivary gland origin had been reported in the English-language literature. We report a new case, which occurred in a 48-year-old woman whose disease had metastasized to regional lymph nodes. She was treated by radical parotidectomy, with sacrifice of the facial nerve, and radical neck dissection. At the 3-year follow-up, she remained free of disease. Our review of the literature suggests that patients with such tumors who undergo aggressive surgery rather than conservative treatment have the best prognosis.

Introduction

Oncocytes are most abundant in the parotid gland; they are also found in other major and minor salivary glands as well as in the larynx, trachea, bronchi, esophagus, nasal mucous membranes, thyroid, parathyroid, pancreas, liver, and stomach. (1) The terms oncocytic carcinoma, oncocytic adenocarcinoma, malignant oncocytoma, and malignant oxyphilic adenoma are synonymous.

Oncocytic carcinoma of salivary gland origin is rare. Bauer and Bauer reported the first case in 1953, (2) and only 39 cases had been reported in the English-language literature through the end of 2002. (3-5) Most cases of oncocytic carcinoma have occurred in the parotid glands, but recent reports have described tumors that involved the submandibular gland and minor glands of the palate, nasal cavity, and ethmoid and maxillary sinuses. (6,7) Malignant oncocytic tumors have also occurred in the thyroid and parathyroid glands, kidney, ovary, nasal cavity, superior mediastihum, lung, and breast. (7) Although focal oncocytic features are seen in a wide variety of salivary neoplasms, both benign and malignant oncocytic neoplasms are extremely rare. (8) In this article, we describe a new case of oncocytic carcinoma that arose in the parotid gland and metastasized to regional lymph nodes.

Case report

A 48-year-old woman was admitted to the ENT clinic with a 3-year history of a painless left preauricular mass. Physical examination revealed a fixed, hard, 6 x 6-cm mass in the left parotid region along with a partial left peripheral facial paralysis. Multiple mobile lymph nodes were palpable on the left side of her neck. Computed tomography demonstrated a 6 x 6-cm solid lesion in the left superficial parotid region and several enlarged lymph nodes in the parotid gland and on the left side of the patient's neck. Systemic physical and laboratory examinations revealed no abnormality. Cytologic examination of a fine-needle aspiration biopsy specimen suggested Warthin's tumor. A second aspiration biopsy specimen was reported as a malignant epithelial tumor. After the patient provided written consent for sacrifice of the facial nerve during a possible radical parotidectomy and neck dissection, we performed a superficial parotidectomy, which leaves residual tumor on the VIIth cranial nerve. The specimen was sent for frozen-section analysis, which revealed the presence of malignant-appearing cells. Therefore, a radical parotidectomy with sacrifice of the facial nerve and a radical neck dissection were performed. Four enlarged lymph nodes between 1 and 3 cm in diameter were found in neck levels 2, 3, and 4. No reconstructive procedure was performed on the massively infiltrated facial nerve.

Histopathologic examination of paraffin-block sections stained with hematoxylin and eosin (H&E) revealed that the tumor had almost completely infiltrated the parotid gland. Nuclear atypia of the tumor cells was not as prominent as it usually is in oncocytomas. Cells with abundant polygonal eosinophilic granular cytoplasm formed nests (figure 1). Mitoses were scattered. The four lymph nodes were positive for metastasis, as they had the same histopathologic features as did the primary tumor (figure 2). Streptavidin-biotinimmunohistochemical study of the parotidectomy and neck dissection specimens was performed on paraffin-block sections by the standard labeled method. The primary antibody was a mouse monoclonal antihuman mitochondrial antibody (MAB 1278; Chemicon International; Temecula, Calif.). Almost all antibody tumor cells showed fine-granular, intense immunoreactivity in the cytoplasm (figure 3). A diagnosis of oncocytic carcinoma was based on these histopathologic and immunohistochemical findings.

[FIGURES 1-3 OMITTED]

Postoperative radiotherapy with 50 Gy to the neck and parotid region was administered, and the patient showed no signs of recurrence or distant metastasis at 3 years of follow-up.

Discussion

Oncocytic adenocarcinomas have been estimated to represent only 5% of all oncocytomas and 0.0005% of all salivary gland tumors. (9) They are characterized by oncocytes with marked cellular atypia, frequent mitoses, destruction of adjacent structures, perineural or vascular invasion, and distant or regional lymph node metastasis. All types of benign or malignant salivary gland tumors may have foci of oncocytic cells, but the oncocytic component usually contains such a small portion that it is unlikely to be confused with oncocytic carcinoma. Tumors with a significant oncocytic component include Warthin's tumor, oncocytoma, and oncocytic carcinoma. (10) Histochemical or electronmicroscopic confirmation of the oncocytic (mitochondrial) nature of the cytoplasm is necessary because cytoplasmic accumulation of smooth endoplasmic reticula, lysosomes, or secretory granules may have a similar appearance. (7) Shintaku and Honda reported that immunohistochemistry with antimitochondrial antibody is highly sensitive and specific for light microscopic identification of mitochondria and superior to routine H&E staining, especially for the detection of isolated oncocytic cells. (11) In our patient, the immunohistochemical study with antimitochondrial antibody revealed intense immunoreactivity of the cytoplasm in the tumor ceils.

Approximately one-third of patients with oncocytic carcinoma of the parotid develop a painful mass or experience facial paralysis. The skin overlying the gland is occasionally discolored or wrinkled. Diagnosis is usually made 1 to 2 years after the onset of disease. (7)

Nakada et al published a review of 27 cases oncocytic carcinoma of the parotid gland. (6) Those patients ranged in age from 30 to 91 years (mean: 58). Local lymph node metastasis occurred in 17 patients (63.0%) and distant metastasis in 7 (25.9%). They reported that distant metastasis appeared to be the most important prognostic feature of oncocytic carcinoma; local lymph node metastasis was not necessarily a critical factor in the overall prognosis. The 3-year recurrence-free survival of our patient, despite the fact that she had had a neck metastasis, is consistent with survival reported by Nakada et al.

Goode and Corio reported nine cases of oncocytic carcinoma of the salivary glands, which they obtained from the files of the Armed Forces Institute of Pathology. (9) Recurrences developed in five of these nine patients (55.6%), four of whom had been treated initially with conservative rather than aggressive surgical intervention. Goode and Corio reported that tumors smaller than 2 cm in diameter appeared to be associated with a better prognosis than those that were larger. It seems clear from their study that patients who undergo more aggressive initial surgery have a significantly better overall prognosis.

Radiation does not appear to favorably alter the biologic behavior of this tumor. (9) Prophylactic neck dissection may be indicated for tumors that are larger than 2 cm in diameter. (9) Further investigation of the prognosis of patients with oncocytic carcinoma of the parotid gland is warranted as more cases are reported.

References

(1.) Chu W, Strawitz JG. Oncocytoma of the parotid gland with malignant change. Arch Surg 1978;113:318-19.

(2.) Bauer WH, Bauer JD. Classification of glandular tumors of the salivary glands. Study of 143 cases. Arch Pathol Lab Med 1953; 55:328-46.

(3.) Ardekian L, Manor R, Peled M, Laufer D. Malignant oncocytoma of the parotid gland: Case report and analysis of the literature. J Oral Maxillofac Surg 1999;57:325-8.

(4.) Alberty J, August C, Stoll W. [Oncocytic neoplasms of the parotid gland. Differential diagnosis, clinical course and review of the literature]. HNO 2001;49:109-17.

(5.) Capone RB, Ha PK, Westra WH, et al. Oncocytic neoplasms of the parotid gland: A 16-year institutional review. Otolaryngol Head Neck Surg 2002; 126:657 62.

(6.) Nakada M, Nishizaki K, Akagi H, et al. Oncocytic carcinoma of the submandibular gland: A case report and literature review. J Oral Pathol Med 1998;27:225-8.

(7.) Ellis GL, Auclair PL. Tumors of the salivary glands. In: Ellis GL, Auclair PL, eds. Atlas of Tumor Pathology. 3rd series, fascicle 17. Washington, D.C.: Armed Forces Institute of Pathology, 1996: 318-24.

(8.) Eneroth CM, Zajicek J. Aspiration biopsy of salivary gland tumors. II. Morphologic studies on smears and histologic sections from oncocytic tumors (45 cases of papillary cystadenoma lymphomatosum and 4 cases of oncocytoma). Acta Cytol 1965; 9:355-61.

(9.) Goode RK, Corio RL. Oncocytic adenocarcinoma of salivary glands. Oral Surg Oral Med Oral Pathol 1988;65:61-6.

(10.) Paulino AF, Huvos AG. Oncocytic and oncocytoid tumors of the salivary glands. Semin Diagn Pathol 1999;16:98-104.

(11.) Shintaku M, Honda T. Identification of oncocytic lesions of salivary glands by anti-mitochondrial immunohistochemistry. Histopathology 1997;31:408-11.

From the Department of Otolaryngology (Dr. Cinar, Dr. Vural, and Dr. Turgut) and the Department of Pathology (Dr. Basak), Sisli Etfal Teaching and Research Hospital, Istanbul, Turkey.

Reprint requests: Ugur Cinar, MD, Korkadi sok. Turizm sitesi, 4 Blok No. 13 Ulus, Istanbul, Turkey 80600. Phone: 90-212-263-1878; fax: 90-212-266-1127; e-mail: u_cinar@yahoo.com
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Article Details
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Title Annotation:Original Article
Author:Turgut, Suat
Publication:Ear, Nose and Throat Journal
Geographic Code:7TURK
Date:Sep 1, 2003
Words:1476
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