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Orbital Relapsing Polychondritis: A Unique Presentation, Complication, and Treatment
Grant H Moore1, Daniel B Rootman, C Nathaniel Roybal
1Department of Oculoplastic Surgery, Jules Stein Eye Institute, UCLA, Los Angeles, CA, U.S.A.
Ophthalmic Plastic and Reconstructive Surgery
|July 30, 2014
Summary
Relapsing polychondritis can cause orbital inflammation. Anakinra effectively treated a patient’s ophthalmic symptoms and prevented Sweet Syndrome recurrence, suggesting its potential for managing these rare complications.
Area of Science:
- Ophthalmology
- Rheumatology
- Immunology
Background:
- Relapsing polychondritis (RP) is a rare systemic autoimmune disease characterized by progressive destruction of cartilaginous structures.
- Ophthalmic manifestations of RP, though uncommon, can lead to significant morbidity.
- Orbital inflammation is a rare but serious complication of relapsing polychondritis.
Observation:
- An 87-year-old male with RP presented with periorbital swelling and erythema, initially treated as orbital cellulitis.
- Initial antibiotic treatment failed, but intravenous methylprednisolone rapidly improved symptoms, suggesting an autoimmune etiology.
- Adalimumab, a TNF-alpha inhibitor, was initiated as a steroid-sparing agent but led to the development of Sweet Syndrome.
Findings:
- Orbital inflammation secondary to relapsing polychondritis was diagnosed.
- Anakinra, an IL-1 receptor antagonist, was initiated after adalimumab-induced Sweet Syndrome.
- Anakinra treatment resulted in significant clinical improvement and prevented recurrence of Sweet Syndrome.
Implications:
- Anakinra may be a valuable therapeutic option for managing ophthalmic manifestations of relapsing polychondritis.
- This case highlights the potential complications of biologic therapies in RP and the utility of anakinra in specific scenarios.
- Further research is warranted to explore the role of IL-1 blockade in RP-associated ocular inflammation.
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