Purpose : To report a case with history of penetrating keratoplasty (PK) for keratoconus that developed acute hydrops in the recipient and donor cornea

Methods : A 46-year-old man, with history of bilateral keratoconus, who had undergone corneal transplantation in his left eye, presented with complaints of sudden visual reduction, photophobia, redness and pain of the left eye.

Results : Review of his clinical course, slit-lamp biomicroscopy, laboratory evaluations including confocal microscopy and ultrasound biomicroscopy revealed acute hydrops in the graft. Second corneal transplantation was done for his left eye and pathologic examination confirmed the diagnosis.

Conclusion : Acute hydrops can occur after PK in patients with keratoconus. Although this condition is not common, it should be considered as a differential diagnosis of graft rejection.