Purpose:The aim of this study was to review the postoperative course and imaging features of 7 eyes that presented with corneal hydrops after Bowman layer (BL) transplantation was performed for advanced keratoconus to determine the potential mechanisms of hydrops formation.Methods:A retrospective analysis was performed of 7 eyes of 5 patients with advanced keratoconus that underwent midstromal BL transplantation at 2 tertiary referral centers and developed acute corneal hydrops on average 64 (±30) months (range 14–104 months) postoperatively. Corneal tomography and anterior segment optical coherence tomography (AS-OCT) images were reviewed to document the postoperative and posthydrops course.Results:For all eyes, the post-BL transplantation course was uneventful until hydrops development. Despite stable postoperative topographies in 5 of 7 eyes, eyes developed hydrops with typical hypodense areas on AS-OCT that were limited to the stromal layers posterior to the BL graft. With AS-OCT (6/7 eyes), 2 eyes showed a break in Descemet membrane, whereas Descemet membrane was intact across the cornea in 2 eyes; in 2 eyes, the images were inconclusive. All patients admitted to continued eye rubbing, and all but 1 had a clinically significant allergy and/or atopic constitution. Most eyes (5/7) showed a relatively quick (visual) recovery within 1 to 4 months after hydrops.Conclusions:Hydrops formation in keratoconic corneas after midstromal BL transplantation may indicate that a break in Descemet membrane is secondary to hydrops development (and not vice versa). With a midstromal BL graft in situ limiting hydrops dimensions, resolution of the hydrops seemed relatively quick with recovery to prehydrops visual acuity in most eyes.