In a 50-year-old man, penetrating keratoplasty was carried out to treat a diffuse silicone keratopathy that developed 2 years after a pars plana vitrectomy combined with an intravitreal silicone injection for a complicated retinal detachment in the only aphakic eye. At 2 weeks after the intravitreal silicone injection, specular microscopy revealed a mild pleomorphism in the corneal endothelium and an endothelial cell loss of 26%. After 5 months, a small silicone drop floated in the anterior chamber and the first signs of diffuse keratopathy were observed. At the same time, specular microscopy revealed severe damage to the endothelial cells and a cell loss of 69%. After the keratoplasty, scanning electron microscopy of the corneal button showed a filamentous structure of the posterior surface of the cornea, with fibroblast-like cells; the endothelial cells were absent.