To report a rare case of a 62-year-old man who presented with bilateral eyelid swelling, chemosis, and hyperemia of the conjunctiva, which wax and waned, being the initial manifestation of relapsing polychondritis.

A 62-year-old man presented with pain and erythematous swelling of the right eyelid for 2 days. There were no other symptoms except chemosis and hyperemia of the conjunctiva. After 1 week, the same symptoms occurred in the left eye, while the lesion of the right eye underwent improvement and aggravation repeatedly. Blood culture tests, Orbital CT, and MRI were performed, but could not confirm a diagnosis. During the follow-up period, erythematous swelling of the left auricle and laryngopharyngitis appeared and erythematous plaques were scattered on the extremities. Punch biopsies were performed; histopathologic examinations concluded to the diagnosis of chondritis. The diagnosis of relapsing polychondritis was confirmed through histologically diagnosed chondritis and repeated ocular symptoms with systemic features.

When patients repeatedly present bilateral eyelid swelling with hyperemia and chemosis of the conjunctiva, relapsing polychondritis should be considered in the differential diagnosis.