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Purpose: To report an unusual fundus autofluorescence aspect in a patient with suspected hydroxychloroquine retinal toxicity. Method: Case report of an unusual presentation of a patient treated for 9 consecutive years with a therapeutically recommended dose of hydroxychloroquine. Result: We report the case of a 53-year-old Caucasian female treated with 400 mg hydroxychloroquine for rheumatoid arthritis over 9 years, currently on methotrexate and folinic acid, who stopped treatment 3 years ago. The cumulative dose is estimated at 1.314 kg. She recently noticed a reduction of vision in both eyes to 0.34 logMAR, with colour vision problems and major distortion in central vision. Fundus autofluorescence revealed minimal foveal pigmentary changes and more pronounced changes in the retina elsewhere. Foveal changes were confirmed by optical coherence tomography in both eyes. The patient did not report any colour perception difficulties or night vision problems and has no family history of any eye condition. Her visual field tested by an optician was full, with some central changes. Conclusion: Retinal toxicity from hydroxychloroquine can present in a different aspect than the commonly known retinal toxicity, and it happens even after years of cessation of the drug. The role of cumulative dose in toxicity is supported in this paper. [ABSTRACT FROM AUTHOR]