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Purpose: To demonstrate that rifabutin-related corneal deposits are localized to the deep stroma using anterior segment optical coherence tomography (OCT) and confocal microscopy. Observations: A 55-year-old male with a history of human immunodeficiency virus (HIV) and disseminated mycobacterium avium complex on rifabutin treatment for 3 years presented with bilateral corneal deposits. Confocal microscopy and anterior segment OCT confirm that rifabutin-related corneal deposits are located in the deep stroma, rather than in the endothelium. Conclusions: And Importance: Rifabutin deposits localize to the deep corneal stroma, and can be seen with both confocal microscopy and anterior segment OCT. Anterior segment OCT is a widely available and easily used diagnostic tool, and can provide utility in the diagnosis of corneal deposits.