Journal List > J Korean Ophthalmol Soc > v.52(12) > 1008962

Jeong and Kim: Relapsing Polychondritis Presenting with Repeated Inflammation of Bilateral Eyelid and Conjunctiva

Abstract

Purpose

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.

Case summary

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 followup 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.

Conclusions

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

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Figure 1.
Orbit CT reveals mild asymmetric swelling of the right eyelid with no evidence of mass-like lesions. Orbit CT of the left eye shows normal findings.
jkos-52-1524f1.tif
Figure 2.
One week after initial treatment, photograph shows normal appearance of the right eyelid and conjunctiva. However, severe erythematous swelling occurred previously on the right eyelid can be seen on the left eyelid.
jkos-52-1524f2.tif
Figure 3.
(A, B) There is no evidence of mass-like lesions in T1 and T2 images. (C, D) Enhanced view shows strong enhancement of left lacrimal area and superior orbital tissue.
jkos-52-1524f3.tif
Figure 4.
Photograph shows recurred erythematous eyelid swelling and conjunctival hyperemia of the right eye. Left eyelid swelling improved from 1 week ago, but swelling and hyperemia are still present. The patient also experienced bilateral hearing loss, sore throat, and fever.
jkos-52-1524f4.tif
Figure 5.
(A) Photograph shows erythematous swelling of the left auricle. Erythematous swelling is localized to the cartilaginous portion of the helix sparing the ear lobe. (B) Photograph shows pea- to-walnut-sized erythematous plaques scattered on the extremities.
jkos-52-1524f5.tif
Figure 6.
Pathological findings of the auricular cartilage (HE staining, ×200). Infiltration of neutrophils and lymphocytes (white arrowhead) are noted in the perichondrium. Infiltration and congestion of dermal perivascular mixed cells (white arrow) are also seen in the soft tissue and perichondrium.
jkos-52-1524f6.tif
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