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Pellucid marginal degeneration of the cornea (1,2) is an idiopathic condition characterized by noninflammatory, nonulcerative thinning of the inferior, peripheral cornea. Occasionally the condition is complicated by acute corneal hydrops (3). We describe a patient with findings suggestive of pellucid marginal corneal degeneration who experienced acute hydrops and spontaneous perforation of the cornea.A retrospective analysis of the case was conducted.A 56-year-old woman was seen with decreased vision in the left eye. Clinical findings, including computerized corneal topography, were suggestive of pellucid marginal degeneration. Two weeks later, the patient had hydrops and spontaneous perforation of the left cornea. Medical workup of the patient and histopathologic and microbiologic studies of the corneal specimen revealed no contributory disease process. The patient underwent emergency tectonic lamellar keratoplasty and is doing well after subsequent optical penetrating keratoplasty.We believe this is the first report of a case of pellucid marginal degeneration of the cornea complicated by hydrops and perforation.