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Fuchs endothelial corneal dystrophy (FECD) is an acquired corneal endotheliopathy and is one of the most common indications for corneal transplantation surgery worldwide. Endothelial keratoplasty (EK) is the most popular form of corneal transplantation for FECD. In standard EK surgery, the patient's corneal endothelium and basement membrane [ie, Descemet membrane (DM)] are first removed, followed by transplantation of donor tissue that comprises allogenic corneal endothelial cells, DM, and corneal stroma of variable thickness. We hypothesized that in lieu of EK, transplantation of acellular DM (ie, Descemet membrane transplantation, DMT) may similarly restore anatomical and functional integrity of the corneal endothelium, by stimulating centripetal migration of peripheral host corneal endothelial cells.A case report of a first-in-human trial of DMT for treatment of FECD is presented.A patient with FECD was successfully treated with DMT. Her preoperative best-corrected Snellen visual acuity (BCVA) was 6/18, central corneal thickness was 603 nm, and central corneal endothelial cell density was unrecordable. By postoperative month 6, her best-corrected Snellen visual acuity had improved to 6/7.5, central corneal thickness was 569 nm, and central corneal endothelial cell density was 889 cells/mm2. She remained stable despite complete cessation of all medications including immunosuppressants. No significant postoperative complications have been encountered.DMT may be effective for treatment of FECD. Achievement of endothelial regeneration without allogenic corneal endothelial cell transplantation and exposure to the attendant risks of graft rejection and chronic immunosuppression represents a significant improvement from the current paradigm of EK.