Objective: To describe a case of bilateral multifocal stromal crystalline keratopathy in the setting of relapsing polychondritis (RP).

Methods: Case report.

Results: We describe a 31-year-old woman who presented with ocular inflammation, bilateral auricular chondritis, and nasal chondritis, meeting the clinical criteria of RP. We illustrate her auricular cartilaginous abnormalities, saddle nose deformity, scleritis, and discrete mid-stromal opacities in both corneas that extend through the central cornea. Uniquely, her opacities feature a marked crystalline component as demonstrated on photography, anterior segment optical coherence tomography, and confocal microscopy.

Conclusions: A central keratopathy is not typically reported in patients with RP. In this case report, we describe a unique diffuse bilateral nummular mid-stromal crystalline keratitis that extends into the central cornea and further define it using multimodal imaging.