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Lacrimal gland swelling is usually due to an inflammatory or neoplastic process. We report an oncocytoma as the cause of lacrimal gland swelling and review the literature. Oncocytoma of the lacrimal gland is extremely rare and has been described only three times before.
Case report
A 72 year old man experienced periodic swelling of his right eyelid over a period of 9 months. For 2 months he complained of vertical diplopia. He had a past medical history of chronic obstructive airways disease and a past ocular history of early cataract, asteroid hyalosis of the vitreous body, and primary open angle glaucoma.
On examination visual acuity of both eyes was 0.50. External examination showed a right hypotropia. There was a 3 mm of proptosis of the right eye. Eye movements in that eye were restricted in all directions. Slit lamp evaluation showed bilateral mild cataract. Intraocular pressures were normal (12 and 10 mm Hg respectively). Funduscopic examination yielded cup:disc ratios of 0.8. Computed tomography (CT) of orbit (Fig 1) showed an extraconally solid space occupying mass in the lacrimal fossa. The tumour displaced the right eye inferomedially. The tumour was thought to arise from the lacrimal gland. A fine needle aspiration cytology demonstrated proliferation of oncocytic cells. The tumour was removed in toto via a lateral orbitotomy. The operation and the postoperative period were uncomplicated.
CT scan showing tumour in the right orbit (arrow). Axial slice shows proptosis of the right eye.
Histological macroscopic description yielded a spherically shaped, partially encapsulated tissue (measuring 2.8 × 3.0 × 1.9 cm). After preparation it had a homogeneous brownish tuberiform aspect. Microscopic examination (Fig 2) showed fibrous tissue with focally pre-existing remnants of lacrimal gland tissue. There was a sharply demarcated tumour composed of tubular structures lined by large cells. These transformed epithelial cells consisted of eosinophilic, granular cytoplasm with round to oval shaped relative small nuclei and nucleoli. No evidence of angioinvasive growth was seen. A diagnosis of a radically removed oncocytoma was made.
Microscopic image showing oncocytic cells in the lacrimal gland.
Postoperative visual acuity of the right eye was reduced because of poor tear secretion confirmed on Schirmer testing and slit lamp examination showed filamentary keratopathy. Lubricating eye drops were prescribed and collagen punctal plugs were inserted in the superior and inferior puncta. Diplopia and the motility disturbance diminished. Follow up to date is 1½12 years and no regrowth has been assessed.
Comment
Oncocytomas (synonyms: oxyphilic adenomas or oncocytic adenomas) are benign, epithelial tumours arising in the ductal cell lining of apocrine glandular structures. In an oncocytoma the oncocytes can form nests, cords, and tubules. Oncocytes can be found among the epithelial cells of various normal organs.1 Oncocytomas occur frequently and have been described in salivary, thyroid, parathyroid, buccal mucosa, breast, kidney, pharynx, and larynx. If they occur near the eye, they are generally located in caruncle, lacrimal sac, accessory lacrimal glands of the conjunctiva and rarely in the lacrimal gland. For unknown reasons oncocytic lesions begin to appear in early adulthood and proliferate with age.2
Most lacrimal gland masses represent inflammatory disease, either responding to antibiotics or anti-inflammatory medication. Various malignant tumours of the lacrimal gland have been described varying from lymphoproliferative disorders to epithelial neoplasms. Approximately 50% of epithelial tumours are benign mixed tumours (pleomorphic adenomas) and about 50% are carcinomas. In rare cases a lacrimal gland mass is found to be an oncocytoma, as described in our case and in three other cases in the literature,3,4 or as an oncocytic carcinoma.4–6
Beskid and Zarzcka3 described a 39 year old woman with an oncocytoma of the lacrimal gland. Riedel et al4 reported a 1.5 year old African girl with an oncocytoma of the lacrimal gland. This is the only case in the literature that reported on an oncocytoma in childhood. Riedel et al4 also reported a 76 year old woman with an oncocytoma of her lacrimal gland, that presented with a 2 month period of swelling of her lacrimal fossa without proptosis. After excision there was no regrowth during a follow up period of 3.5 years. Proliferations of oncocytic cells can also show malignant features and become malignant oncocytomas. A 58 year old man reported by Riedel4 had a malignant oncocytoma (synonym: oxyphilic adenocarcinoma) of the lacrimal gland. He had a 3 month history of proptosis of his eye and diplopia. On presentation the tumour had intracranial extension. Despite radical resection and postoperative radiation therapy, he died 6 months later from liver metastases. Dorello et al5 described a similar case of a 59 year old patient with an oncocytic carcinoma of the lacrimal gland with intracranial extension. The patient died approximately 2 years after development of his clinical symptoms (exophthalmos and diplopia), despite orbital exenteration and radiation therapy. A 81 year old woman reported by Biggs6 had a 6 month history of proptosis due to an oncocytic adenocarcinoma of the lacrimal gland. No follow up information was given.
In summary, a 72 year old man was found to have an oncocytoma of the lacrimal gland. Oncocytoma has to be added to the differential diagnosis of lacrimal gland swelling.