Complication of Ectopic Lacrimal Gland Tumor on Long-term Follow-up: A Case Report.

Dear Editor, Ectopic lacrimal gland tissue is reported to be located at epibulbar conjunctiva, other locations including orbital, eyelid, intraocular, lacrimal sac, and nasal mucosa sites [1]. Although ectopic lacrimal glands in the ciliary body are quite rare, there have been more than 10 cases reported worldwide [2,3]. Although accompanying proptosis at the time of diagnosis has been frequently reported, studies on long-term follow-up outcomes and complications are limited. Herein, we report a single case of ectopic lacrimal gland on ciliary body, resulting in secondary glaucoma during 20 years of follow-up. Informed consent for publication of the research details and clinical images was obtained informed consent from the patient. A previously healthy 10-month-old female patient presented with right upper eyelid swelling and redness. And she was admitted to an emergency room in 2003. B-scan ultrasonography and orbit computed tomography demonstrated right orbital cellulitis and a round-shaped, 0.8-cm ciliary body mass. Fine needle aspiration and incisional biopsy were done, which revealed ectopic lacrimal gland tumor in histology. The size of residual mass gradually increased and there was no sign of malignant transformation during 17 years of follow-up period. The patient was transferred to the glaucoma clinic due to intermittent high intraocular pressure and corresponding ocular pain of the right eye once or twice a month in 2020. Thus, she started dorzolamide 2%/timolol maleate 0.5% fixed combination (Cosopt, Merck & Co Inc). No other abnormal findings were observed on slit-lamp examination. On January 10, 2021, she was admitted to an emergency room due to ocular pain and decreased vision in the right eye that started a day ago. Best-corrected visual acuity was 2 / 20 and intraocular pressure (IOP) measured by Goldman applanation tonometer was 44 mmHg. Slit-lamp examination showed corneal edema and moderate anterior chamber inflammation (cell grade from +2 to +3). Anterior chamber was deep enough and there was no peripheral angle closure. Pupil was fixed and fundus was normal in the right eye. The left eye was normal. Under the impression of secondary glaucoma associated with uveitis, she was treated with additional oral acetazolamide, topical prednisolone acetate 1% (Prednilone, Daewoo Pharm), brimonidine 0.15% (Alphagan–P, Allergan Inc) and intravenous mannitol injection. IOP was in the normal range after 3 days. Although she used Cosopt and Alphagan prophylactically, there were two more additional acute glaucoma attacks in 2021. Slit-lamp examination revealed new cystic lesion in superotemporal iris in March 2, 2022 (Fig. 1A). On July 6, 2022, the cystic lesion became larger than 4 months before (Fig. 1B), but there was no other abnormal sign such as increased IOP or anterior chamber inflammation. On July 29, 2022, she was admitted to emergency room with a third attack, presented with decreased visual acuity and 33 mmHg of IOP in the right eye. The size of ciliary body mass was slightly decreased but there was flare reacReceived: March 17, 2023 Final revision: April 21, 2023 Accepted: May 30, 2023