We present a case of unilateral acute hydrops that developed in 25-year-old man 6 years after bilateral laser in situ keratomileusis (LASIK) for the correction of 2.75 diopters (D) of myopia. Preoperative corneal thickness using ultrasound pachymetry were 556 μm in the right eye and 554 μm in the left eye. Preoperative corneal topography of both eyes showed an asymmetric bow-tie pattern with inferior steepening. Forty-seven months after LASIK, the left eye developed astigmatism and enhancement excimer ablation was performed. Sixteen months after enhancement, a definite sign of keratectasia was detected in the left eye. Twenty-six months after enhancement, acute hydrops occurred in the left eye. The interface of the LASIK wound was separated and filled with aqueous humor. Penetrating keratoplasty was performed to avoid perforation. The keratocytes at the edge between the peripheral flap and remaining stroma showed mixoid degeneration and edematous change suggesting dying cells.