Purpose: To present a case with unusual epithelial basement membrane detachment.Methods: A 29-year-old female presented with blurred-vision and pain in left-eye for 2 months. Slit-lamp showed almost total bullous epithelial detachment with subepithelial fluid, and intraocular pressure rise. Recurrences were observed, despite repeated epithelial debridement and topical medication.Results: Decrease in corneal endothelial cell density and changes resembling cytomegalovirus (CMV) keratitis were noted on specular microscopy. PCR for CMV came negative; however, we had success with topical gancyclovir and steroids, in addition to topical antiglaucomatous agents and artificial tears. During the following 12 months, no recurrence was observed.Conclusion: According to her clinic and response to medication, we believe that this case represents an atypical form of CMV endotheliitis.