Clinical case of acute hydrops treatment using subtotal penetrating keratoplasty (PK) is presented. The diagnosis of acute hydrops was based on clinical and functional evaluations including optical coherent tomography (OCT). A part of diseased cornea was removed and examined under a light microscope. These studies revealed morphological changes in almost all corneal layers. OCT and histology demonstrated that PK was indicated to this patient. Recent literature data on the epidemiology and pathogenesis of acute hydrops are presented. This rare disease results from tears in the Descemet’s membrane that allow aqueous humor to enter the stroma. Current treatment is aimed to suppress corneal inflammation, restore endothelium and Descemet’s membrane integrity and drain stromal cysts to optimize cornea healing. In this case, subtotal PK was performed due to the significant corneal thinning and a high risk of its melting. «Material for corneal graft» (iLab, Moscow, Russia) was used as a donor material.