Purpose: To present a rare case of microsporidial keratoconjunctivitis in an otherwise healthy contact lens wearer without human immunodeficiency virus infection who responded to treatment with systemic albendazole and topical fumagillin.
Design: Interventional case report.
Method: A cornea epithelial scraping from a man with unilateral keratoconjunctivitis previously treated with topical steroids was evaluated by modified trichome staining.
Main outcome measures: The patient was evaluated for his symptoms, visual acuity, clinical observations, and pathologic examination of corneal scrapes.
Results: Modified trichome staining of an epithelial corneal scraping revealed pinkish to red organisms characteristic of microsporidia. Results of a human immunodeficiency virus (HIV) enzyme-linked immunosorbent assay test were negative. The symptoms of ocular discomfort and clinical signs of keratoconjunctivitis resolved after 2 months of treatment with albendazole and topical fumagillin.
Conclusions: Ocular infection with microsporidia, although classically occurring in patients with HIV infection, may occur rarely in healthy individuals, especially if previously treated with systemic immune suppression or topical steroids. Microsporidial keratoconjunctivitis should be considered in the differential diagnosis of a contact lens wearer with atypical multifocal diffuse epithelial keratitis.