This article has Open Peer Review reports available.
Oncocytic carcinoma of the parotid gland with late cervical lymph node metastases: a case report
© Gallego et al; licensee BioMed Central Ltd. 2011
Received: 15 March 2010
Accepted: 17 January 2011
Published: 17 January 2011
Oncocytic carcinoma is a rare proliferation of cytomorphologically malignant oncocytes mainly found in glandular tissue, accounting for 0.5% of all epithelial salivary gland malignancies and 0.18% of all epithelial salivary gland tumors.
We report a case of oncocytic carcinoma arising in the parotid gland of a 65-year-old Caucasian man. Our patient initially underwent left superficial parotidectomy, including the removal of the mass. A close follow-up was made, and four years after first surgery cervical lymph node metastases were confirmed. Therefore, a complete parotidectomy and radical neck dissections were performed. There were no complications and no sign of recurrence after six months of follow-up.
Oncocytic carcinoma is an extremely rare malignancy in the salivary glands. Prophylactic neck dissection may be indicated for tumors larger than 2 cm in diameter (our patient's tumor was 2.5 cm at its greatest diameter). The clinical course of our patient, with the appearance of cervical lymph node metastases after four years of follow-up, supports this approach. Further investigation of the prognosis and correct treatment of patients with oncocytic carcinoma are required as more cases are reported.
Oncocytic carcinoma (OC) is an unusual proliferation of cytomorphologically malignant oncocytes and adenocarcinomatous architecture phenotypes mainly found in glandular tissue . The terms oncocytic carcinoma, oncocytic adenocarcinoma, malignant oncocytoma and malignant oxyphilic adenoma are synonymous. Its malignant nature is distinguished from oncocytoma by abnormal morphological features and infiltrative growth . Necrosis, peri-neural spread, pleomorphism, intra-vascular invasion, and distant metastasis to the cervical lymph nodes, kidneys, lungs, and mediastinum are the main features of this high-grade malignant tumor .
This tumor represents 5% of all oncocytic salivary gland neoplasms and less than 1% of all salivary gland tumors . OC may occur in many sites in addition to the salivary glands, including the nasal and thoracic cavities, ovary, kidney, thyroid gland, breast and parathyroid. The majority of OC cases have occurred in the parotid glands, but tumors that involved the submandibular gland and minor glands of the palate have also been described .
In this case report we describe a case of OC that arose in our patient's parotid gland and presented regional lymph node metastases four years after first surgery.
A month later, our patient underwent left superficial parotidectomy, including the removal of the mass. Because of clear infiltration of the tumor, the buccal branch of the facial nerve was resected. Macroscopically, the tumor was a well-circumscribed, firm, grey-brown, ovoid nodule measuring 2.5 cm in diameter. Microscopic examination revealed that the neoplasm had replaced a wide area of the parotid gland and peri-neural invasion was found (Figure 1B). The neoplastic elements were large, round or polyhedral cells and were arranged in solid sheets, islands and cords. The cytoplasm was abundant, eosinophilic and finely granular, characteristics indicative of oncocytes (Figure 1C). A diagnosis of OC was based on these histopathological findings and in particular on the invasive growth pattern.
OC is an extremely rare malignancy in salivary glands, accounting for just 0.5% of all epithelial salivary gland malignancies and 0.18% of all epithelial salivary gland tumors . Criteria for the diagnosis of malignancy in salivary oncocytic tumors include: (1) local lymph node metastasis; (2) distant metastasis; (3) peri-neural, vascular or lymphatic invasion; (4) frequent mitoses and cellular pleomorphism with extensive invasion and destruction of adjacent structures . This tumor is predominantly composed of round or polyhedral cells arranged in small clusters and occasional solid sheets. Cells have abundant eosinophilic cytoplasm, a result of excessive numbers of mitochondria . Although not commonly used, it has been reported that anti-mitochondrial antibody is highly specific and sensitive to confirm the oncocytic nature of the granular cytoplasm . FNAC is less sensitive for oncocytic neoplasms, perhaps due to the rarity of these tumors and diagnostic pitfalls previously associated with FNAC (for example, sampling errors and overinterpretation of paucicellular specimens) .
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 . In our case, there was no facial paralysis or discoloration of the overlying skin. The average age of patients has been estimated to be about 60 years, with a male predominance [1, 8].
As with other high-grade salivary gland neoplasms, aggressive surgical intervention is indicated. Therapy for OCs consists of surgical intervention, and total parotidectomy with preservation of the facial nerve whenever possible. The efficacy of radiotherapy is unclear. Radiation does not appear to favorably alter the biological behavior of this tumor. In the case reported, intra-operative frozen sections of the tumor would have been indicated for pathological diagnosis and would have altered the surgical decision. FNAC alone did not determine a correct diagnosis and the first surgical management was inadequate.
A number of studies have reported multiple recurrences of this tumor and regional or distant metastases [2, 6, 8–10]. The cervical lymph nodes may be affected as with other malignant tumors of the parotid gland, but prophylactic neck dissection must be considered individually in the absence of consensus in the literature. Nakada et al. published a review of 28 cases of oncocytic carcinoma of the parotid gland . They concluded 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. Distant metastasis sites include the lung, liver, kidney, mediastinum, thyroid gland and bone.
Prophylactic neck dissection may be indicated for tumors larger than 2 cm in diameter  (our patient's tumor was 2.5 cm at its greatest diameter). This likely indicates a worse prognosis. The clinical course of our case, with the appearance of cervical lymph node metastases after four years of follow-up, supports the approach of Goode and Corio .
The prognosis of oncocytic carcinomas is not well known because of their low incidence. Patients with malignant oncocytoma appear to have good short-term survival, but poor long-term survival . The average survival period has been estimated at 3.8 years with metastasizing tumors . Further investigation of the prognosis and correct treatment of patients with OC are required as more cases are reported.
OC is an extremely rare malignancy in salivary glands, and standard treatment and prognosis is unclear. Clinicians should perform a careful follow-up, as distant metastasis appeared to be the most important prognostic feature.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- Ellis GL, Auclair PL: Tumors of the salivary glands. Atlas of Tumor Pathology. 2008, Washington, DC: Armed Forces Institute of Pathology, 356-363. fascicle 9, 4Google Scholar
- Capone RB, Ha PK, Westra WH, Pilkington TM, Sciubba JJ, Koch WM, Cummings CW: Oncocytic neoplasms of the parotid gland: a 16-year institutional review. Otolaryngol Head Neck Surg. 2002, 126: 657-662. 10.1067/mhn.2002.124437.View ArticlePubMedGoogle Scholar
- Barnes L, Eveson JW, Reichart P, Sidransky D: Pathology and genetics of head and neck tumors. World Health Organization Classification of Tumors. 2005, Lyon, France: WHO, 235-Google Scholar
- Guclu E, Oghan F, Ozturk O, Alper M, Egeli E: A rare malignancy of the parotid gland: oncocytic carcinoma. Eur Arch Otorhinolaryngol. 2005, 262: 567-569. 10.1007/s00405-004-0871-4.View ArticlePubMedGoogle Scholar
- Giordano G, Gabrielli M, Gnetti L, Ferri T: Oncocytic carcinoma of parotid gland: a case report with clinical, immunohistochemical and ultrastructural features. World J Surg Oncol. 2006, 4: 54-10.1186/1477-7819-4-54.View ArticlePubMedPubMed CentralGoogle Scholar
- Gray SR, Cornog JL, Seo IS: Oncocytic neoplasms of salivary glands: a report of fifteen cases including two malignant oncocytomas. Cancer. 1976, 38: 1306-1317. 10.1002/1097-0142(197609)38:3<1306::AID-CNCR2820380333>3.0.CO;2-A.View ArticlePubMedGoogle Scholar
- Shintaku M, Honda T: Identification of oncocytic lesions of salivary glands by anti-mitochondrial immunohistochemistry. Histopathology. 1997, 31: 408-411. 10.1046/j.1365-2559.1997.2870891.x.View ArticlePubMedGoogle Scholar
- Nakada M, Nishizaki K, Akagi H, Masuda Y, Yoshino T: Oncocytic carcinoma of the submandibular gland: a case report and literature review. J Oral Pathol Med. 1998, 27: 225-228. 10.1111/j.1600-0714.1998.tb01946.x.View ArticlePubMedGoogle Scholar
- Cinar U, Vural C, Basak T, Turgut S: Oncocytic carcinoma of the parotid gland: report of a new case. Ear Nose Throat J. 2003, 82: 699-701.PubMedGoogle Scholar
- Goode RK, Corio RL: Oncocytic adenocarcinoma of salivary glands. Oral Surg Oral Med Oral Pathol. 1988, 65: 61-66. 10.1016/0030-4220(88)90193-4.View ArticlePubMedGoogle Scholar
- Ardekian L, Manor R, Peled M, Laufer D: Oncocytic neoplasms of the parotid gland: a 16-year institutional review. J Oral Maxillofac Surg. 1999, 57: 325-328. 10.1016/S0278-2391(99)90682-1.View ArticlePubMedGoogle Scholar
- Brandwein MS, Huvos AG: Oncocytic tumors of major salivary glands. A study of 68 cases with follow-up of 44 patients. Am J Surg Pathol. 1991, 15: 514-528. 10.1097/00000478-199106000-00002.View ArticlePubMedGoogle Scholar
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.