Introduction: To report a case of unilateral corneal perforation due to isolated ocular lichen planus. Methods: Interventional case report. Informed consent by the patient was obtained to publish clinical images. A 64-year-old male presented with severe vision loss and a 2-week history of corneal perforation treated with penetrating keratoplasty in the left eye. He had a longstanding diagnosis of severe chronic dry eye disease. On the initial assessment a visual acuity of 20/50 in the RE and HM perception in the left eye were documented. Biomicroscopy revealed subepithelial fibrosis on the tarsal conjunctiva and clinical signs of severe dry eye disease in both eyes. A clear corneal button and a white cataract were observed in the left eye. No other skin or mucosal lesions were observed. Results: An excisional biopsy of the bulbar conjunctiva was performed under topical anesthesia. Direct immunofluorescence analysis revealed a linear deposit of fibrinogen in the basement membrane consistent with ocular lichen planus. Clinical improvement was achieved using aggressive topical lubrication, corneal epithelial regenerators, topical tacrolimus, and immunosuppressive therapy with systemic corticosteroids and cyclophosphamide. Conclusion: Isolated ocular lichen planus is an extremely infrequent presentation of lichen planus often indistinguishable from other cicatricial conjunctivitis. Corneal perforation is a severe complication associated with severe dry eye, not previously reported with ocular lichen planus. An adequate clinical assessment and histopathologic diagnosis are crucial to lead prompt treatment and prevent sight-threatening complications.
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