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To report an unusual case of hydroxychloroquine toxicity after short-term therapy.Observational case report.A 56-year-old woman presented to the Ophthalmology Clinic at Walter Reed Army Medical Center (WRAMC) with a 6-month history of gradually decreasing vision in both eyes. The patient had been taking hydroxychloroquine for the preceding 48 months for the treatment of rheumatoid arthritis. Examination of the posterior segment revealed bilateral “bull's eye” macular lesions. Fundus autofluorescence revealed hyperfluorescence of well-defined bull's eye lesions in both eyes. Optical coherence tomography revealed corresponding parafoveal atrophy with a loss of the retinal inner segment/outer segment junction. Humphrey visual field 10-2 white showed significant central and paracentral defects with a generalized depression. The patient was on a standard dose of 400 mg daily, which was above her ideal dose. The patient had no history of kidney or liver dysfunction. There were no known risk factors but there were several possible confounding factors. The patient was started on high-dose nabumetone, a nonsteroidal antiinflammatory drug, at the same time she was started on hydroxychloroquine. She also reported taking occasional ibuprofen.Retinal toxicity from chloroquine has been recognized for decades with later reports showing retinopathy from long-term hydroxychloroquine (Plaquenil) use for the treatment of antiinflammatory diseases. Hydroxychloroquine is now widely used and retinal toxicity is relatively uncommon. However, it can cause serious vision loss and is usually irreversible. The risk of hydroxychloroquine toxicity rises to nearly 1% with a total cumulative dose of 1,000 g, which is ∼5 years to 7 years of normal use. Toxicity is rare under this dose. For this reason, the American Academy of Ophthalmology has revised its recommendations such that annual screenings begin 5 years after therapy with hydroxychloroquine has begun unless there are known risk factors. This case report confirms the need for a baseline examination and annual ophthalmologic screening for patients taking hydroxychloroquine at a dose higher than the recommended dosage. It is also reasonable to consider annual examinations in patients taking high-dose nonsteroidal antiinflammatory drugs from the initiation of the medication.