A 45-year-old man was diagnosed to have alkaptonuria after his orthopedist observed joint discoloration during knee arthroplasty. He was referred to Dokuz Eylul University, Department of Ophthalmology, for evaluation. On examination, his visual acuity was 20/20 in each eye. Slit-lamp examination revealed brownish pigmented lesions in both the nasal and temporal aspects of the sclera in the interpalpebral region (Osler’s sign) bilaterally. Fundus examination was unremarkable in both eyes.