Retrocorneal membranes can occur after trauma or surgery and remain as a therapeutic problem in graft failure after penetrating keratoplasty. We report for the first time the finding of a retrocorneal membrane in Descemet stripping automated endothelial keratoplasty (DSAEK) in a young woman who underwent Baerveldt shunt implantation for iridocorneal endothelial (ICE) syndrome. Clinical examination, preoperative and postoperative slit lamp-adapted optical coherence tomography of the anterior segment, and immunohistochemical staining and electron microscopic analysis of the stripped Descemet membrane were performed. A 25-year-old woman presented with decline of visual acuity because of the formation of a retrocorneal membrane after Baerveldt shunt implantation for ICE syndrome. Visual acuity improved after DSAEK from 20/200 to 20/63, whereas intraocular pressure did not change in the 6-month postoperative follow-up. Histological and ultrastructural analysis showed a massive retrocorneal membrane. Immunohistopositivity for α-smooth muscle actin, cytokeratin 7, and pan-cytokeratin suggests a metaplastic endothelial origin of this membrane. The exact origin of the retrocorneal membrane found in this case in ICE syndrome after Baerveldt shunt implantation cannot be clarified, but DSAEK is an effective treatment to improve visual acuity in this situation.