Introduction

Oncocytic carcinomas are rare malignant salivary gland tumor most commonly involving major salivary glands. They are epithelial tumors defined by the presence of abundant eosinophilic granular cytoplasm due to high mitochondrial content. Although most commonly identified in the major salivary glands, oncocytic carcinomas may also arise in minor salivary gland tissue in anatomic subsites of the head and neck, including the paranasal sinuses, nasolacrimal system, and nasopharynx. They can also arise in other organs, including the kidney, breast, prostate gland (1).

Case Report

A 65 year old gentleman presented with right nasal blockage since 5 months in the outpatient department of head and neck surgical oncology, Malabar cancer centre, Thalassery. Clinical history revealed epistaxis of 4-5 episodes since 2 months and anosmia. He had hypertension along with coronary artery disease for which he underwent percutaneous transluminal coronary angioplasty 4 years back. On clinical examination there was no proptosis, loss of visual acuity, cheek swelling, headache, and eye movements were normal. Oral cavity and ophthalmic examination were unremarkable. Nasal Endoscopy revealed a tumour in the right nasal cavity, extending to middle meatus involving middle turbinate, medial wall of maxilla, inferior turbinate extending to sphenoethmoidal recess and eroding posterior part of septum. MRI, head and neck showed a polypoidal 46 x 39 x 27 mm lesion involving right nasal cavity infiltrating the nasal septum, eroding medial wall of maxilla with suspicious erosion of cribriform plate with no involvement of dura (Figure 1,2). CT Thorax and abdomen done for metastatic workup was normal.

Figure 1: A Polypoidal 46 x 39 x 27 mm lesion seen involving right nasal cavity. Infiltrating the nasal septum, eroding medial wall of maxilla, suspicious erosion of cribriform plate, dura appears free.

Figure 2: Mucosal thickening with fluid seen within the right maxillary sinus, ethmoid sinus, right frontal sinus and sphenoid sinuses.

An endoscopic biopsy was performed which showed sheets of large cells with abundant eosinophilic granular cytoplasm. The differential diagnosis considered were oncocytic carcinoma, secretory carcinoma and mucoepidermoid carcinoma with oncocytic change. Endoscopic craniofacial resection of sinonasal mass was done and margins were cleared laterally up to lamina papyracea, medially up to nasal septum, inferiorly up to the mucosa of floor of nasal cavity and superiorly to the cribriform plate. Skull base defect were repaired with fascia lata, fibrin glue and nasoseptal flap. The margins and the excised tumor were sent for histopathological examination. The patient received adjuvant radiotherapy and he is under regular follow up.

Microscopic examination revealed an infiltrative neoplasm with tumor cells arranged in sheets, nests and lobules. The individual tumour cells (oncocytes) were large in size with hyperchromatic, pleomorphic nuclei and have prominent nucleoli with eosinophilic granular cytoplasm. Tumor showed mitotic activity of approx. 18-20/10hpf. All the resection margins were found free of tumor.

Figure 3: H and E stained section showing oncocytic cells arranged in sheets [4x(A), 40x(B)]

Discussion

Oncocytic carcinoma of salivary gland origin is an extremely rare tumor composed of malignant oncocytes showing infiltrative qualities, including local invasion, regional or distant metastases(2). Most of the case are reported in major salivary glands. Only a few cases have been reported in nasal cavity. Compared with oncocytomas arising from major salivary glands, those that arise elsewhere from minor salivary glands tend to be more locally aggressive with greater malignant potential, classified as oncocytic carcinomas.(3)

Most of the reported cases of sinonasal oncocytic carcinomas have a male predilection and age>60 years.The clinical presentation of this case is in concordance with the previously reported cases in English literature. Owing to the granular cytoplasm of tumor cells, the histopathological differentials considered were oncocytic carcinoma, oncocytic subtype of mucoepidermoid carcinoma and secretory carcinoma with oncocytic changes. Mucous cells were not identified which ruled out oncocytic subtype of mucoepidermoid carcinoma. Also immunohistochemistry for mammaglobulin was negative in this case.

Sinonasal oncocytic carcinomas are more likely to be malignant compared to that occurring in major salivary glands with more frequent local invasion and recurrence. Regarding treatment of sinonasal oncocytic carcinomas, several surgical approaches have been reported in literature. Surgical excision is the main modality of treatment. The major limitation of reported cases of sinonasal oncocytic carcinomas in literature is the lack of information regarding status of resection margins. Endoscopic approach offers easy access to both the nasal cavity and nasolacrimal apparatus and provide excellent visualization. The success of endoscopic approach depends on experience of the surgical team and optimal preoperative imaging.

The role of adjuvant radiotherapy in preventing recurrence is questioned with the suggestion that these tumors are radioresistant.(12) However, when the disease is locally advanced, or with regional and distant metastasis, adjuvant radiotherapy can be a useful approach to prevent recurrence and spread.(17) Also, regarding the utility of systemic chemotherapy for treating sinonasal oncocytic carcinomas there is insufficient information in the literature.(18) Finally, a diagnosis of sinonasal oncocytic carcinoma warrants careful monitoring and follow-up, as it is locally destructive and capable of metastasis and multiple recurrences.(19)

Conclusion

Owing to the rarity of these tumors, there is no standard management approach, with several different surgical techniques reported in the literature, including both the open and endoscopic approaches, in addition to variable use of radiotherapy. Newer technologies may also help the surgeon to obtain clear surgical margins like fluorescence guided surgery will better show the limits of tissue invasion and thus help to define the surgical margins.

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