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Landolt ring-shaped epithelial keratopathy: a novel clinical entity of the cornea. IMPORTANCE: Landolt ring-shaped epithelial keratopathy is a newly identified disease with vesicular changes of unknown origin in the epithelial cells. OBSERVATIONS: Eleven Japanese patients with specific epithelial lesions that resembled a Landolt ring in the corneal epithelium were assessed. The main symptoms of Landolt ring-shaped epithelial keratopathy are foreign-body sensation and photophobia. Slitlamp examination indicates that Landolt ring-shaped lesions located only in the corneal epithelium are randomly distributed and occur unilaterally, bilaterally, or asymmetrically. Small lesions are sometimes connected to each other to form a large Landolt ring in a fractal pattern. Confocal microscopy reveals that the Landolt ring lesions are vesicular changes in the epithelial cells from the basal cell layer to the superficial cell layer without inflammation. The lesions form for weeks to months with sporadic exacerbations and natural remissions with or without treatments. The outcomes do not include epithelial disorders in the affected corneas or visual disruptions. CONCLUSIONS AND RELEVANCE: We describe a new disease entity of unknown origin referred to as Landolt ring-shaped epithelial keratopathy, which is usually bilateral and characterized by specific Landolt ring-shaped focal epithelial lesions with vesicular changes only in the epithelial cells. The disorder has an insidious onset and self-limiting nature despite treatment and should be included in the differential diagnosis of corneal epithelial disorders.