Purpose: To report a peculiar case of adult-onset buphthalmos.
Methods: Review of the medical record of a 24-year-old patient with Down syndrome who developed buphthalmos in the left eye after corneal transplantation for keratoconus.
Results: In the next 2 years after surgery, the operated eye evolved with chronic anterior uveitis that led to progressive peripheral synechiae, oscillating intraocular pressure, cataract, graft failure, and buphthalmos.
Conclusions: The finding of buphthalmos of adult-onset in Down syndrome raises important issues concerning the classification of this eye condition and the responsibility for detecting and monitoring eyes with glaucoma in individuals with chromosome 21 trisomy.