Abstract
We report a case of nitisinone-induced keratopathy in a 52-year-old male patient with Alkaptonuria (AKU) disease in Jordan. The patient presented with slight drop of left eye vision for the last four weeks, associated with a foreign body sensation in his left eye. On examination, it was found that his left eye vision was reduced to 6/12 and there were left dendrite-like corneal opacities in the left eye. He was given 10 mg of nitisinone per day, two years before his visual complaint. Three months following discontinuation of nitisinone, his visual acuity and corneal opacities disappeared completely. This is the fifth reported case of nitisinone-induced keratopathy, due to secondary tyrosinaemia in alkaptonuria.