Journal List > J Korean Ophthalmol Soc > v.48(12) > 1007995

Yim, Oum, and Park: A Case of Relapsing Polychondritis Complicated with Chorioretinitis without Scleritis

Abstract

Purpose

To report a case of relapsing polychondritis complicated with chorioretinitis without scleritis.

Case summary

A 35-year-old man who has been previously managed for relapsing polychondritis visited our the clinic because of blurred vision in his both eyes which had developed one month earlier. Although the corrected visual acuity was 1.0 in both eyes, Slit lamp examination of both eyes showed findings of anterior uveitis. Fundus examination showed a whitish-yellow lesion around the posterior pole in both eyes. He had bilateral auricular chondritis, saddle nose deformity, and respiratory difficult. The patient was treated with systemic steroid and prescribed topical 1% prednisolone acetate four times daily in both eyes. Four months later, fundus examination of both eyes showed improvement of chorioretinal lesion, but visual acuity had decreased constantly. Nine months later, chorioretinal lesion resolved and visual acuity improved in both eyes.

References

1. Jaksch-Wartenhost R. Poly chon dropathia. Wien Arch F Inn Med. 1923; 6:93–100.
2. Gilliland BC. Relapsing polychondritis. In : Kasper DL, Braunwald E, Fauci AS, editors. Harrison's Principles of Internal Medicine. revised ed.New York: McGraw-Hill;2005. v. 2. chap. 308.
3. Magargal LE, Donoso LA, Goldberg RE, et al. Ocular manifestations of relapsing polychondritis. Retina. 1981; 1:96–9.
crossref
4. Isaak BL, Liesegang TJ, Michet CJ Jr. Ocular and systemic findings in relapsing polychondritis. Ophthalmology. 1986; 93:681–9.
crossref
5. Choi YS, Yim HB, Kim KB. A Case of Episcleritis with Relapsing Polychondritis. J Korean Ophthalmol Soc. 2002; 43:626–30.
6. Joo SH, Choe JK. A Case of Posterior Scleritis Associated with Relapsing Polychondritis. J Korean Ophthalmol Soc. 1989; 30:665–70.
7. McAdam LP, O'Hanlan MA, Bluestone R, Pearson CM. Relapsing polychondritis: prospective study of 23 patients and a review of the literature. Medicine. 1976; 55:193–215.
8. Damiani J, Levine H. Relapsing polychondritis: Report of 10 cases. Layngoscope. 1979; 89:929–46.
9. Letko E, Zafirakis P, Baltatzis S, et al. Relapsing polychondritis: A Clinical review. Semin in Arthritis Rheum. 2002; 31:384–95.
crossref
10. Kaye RL, Sones DA. Relapsing polychondritis: Clinical and Pathologic Features in Fourteen Cases. Ann Intern Med. 1964; 60:653–64.
11. Trentham DE, Le CH. Relapsing polychondritis. Ann Intern Med. 1998; 129:114–22.
crossref
12. Mohsenifar Z, Tashkin DP, Carson SA, Bellamy PE. Pulmonary function in patients with relapsing polychondritis. Chest. 1982; 81:711–7.
crossref
13. Peebo BB, Peebo M, Frennesson C. Relapsing polychondritis: a rare disease with varying symptoms. Acta Ophthalmol Scand. 2004; 82:472–5.
crossref
14. Mckay DA, Watson PG, Lyne AJ. Relapsing polychondritis and eye disease. Br J Ophthalmol. 1974; 58:600–5.
crossref
15. Hoang-Xuan T, Foster CS, Rice BA. Scleritis in relapsing polychondritis. Ophthalmology. 1990; 97:892–8.
crossref
16. Anderson NG, Valenzuela EG, Martin DF. Hypopyon Uveitis and Relapsing polychondritis: A Report of 2 Patients and Review of Autoimmune Hypopyon Uveitis. Ophthalmology. 2004; 111:1251–4.
17. Bhagat B, Green RL, Feldon SE, Lim JI. Exudative retinal detachment in relapsing polychondritis. Am J Ophthalmol. 2001; 108:1156–9.
crossref
18. Anderson B Sr. Ocular lesions in relapsing polychondritis and other rheumatoid syndromes. Am J Ophthalmol. 1967; 64:35–50.
crossref
19. Bergaust B, Abrahamsen AM. Relapsing polychondritis: report of a case presenting multiple ocular complications. Acta Ophthalmol. 1969; 47:174–81.
20. Michelson JB. Melting corneas with collapsing nose. Surv Ophthalmol. 1984; 29:148–54.
crossref
21. Matoba A, Plager S, Barber J, McCulley JP. Keratitis in relapsing polychondritis. Ann Ophthalmol. 1984; 16:367–70.
22. Sundaram MB, Rajput AH. Nervous system complications of relapsing polychondritis. Neurology. 1983; 33:513–5.
crossref
23. Killian PJ, Susac J, Lawless OJ. Optic neuropathy in relapsing polychondritis. JAMA. 1978; 239:49–50.
crossref

Figure 1.
(A) Photographs of both external ears show auricular cartilage deformity. (B) The photograph of nose shows saddle nose deformity resulting from loss of nasal cartilage. (C) Photomicrograph of biopsy specimen from the ear. Degenerating changes in the marginal cartilage and mononuclear inflammatory cells and fibroblasts are seen replacing the destroyed cartilage from the edge of cartilaginous plate (hematoxylin-eosin, *200). (D) Head and neck CT reveals narrowing of the trachea.
jkos-48-1716f1.tif
Figure 2.
Fundus photographs show two 1 disc diameter-sized whitish yellow retinal infiltrates temporal and inferior to the macula, respectively in the right eye (A), and 2 disc diameter-sized whitish yellow retinal infiltrate temporal to the macula in the left eye (B). The horizontal OCT image showed detachment of the neurosensory retina nasally and temporally in the right eye (C), and shallow detachment of the neurosensory retina and focal RPE detachment nasally in the left eye (D). The early phase fluorescein angiogram showed blockage of choroidal fluorescence on retinal infiltrates in the right (E) and left (F) eyes. The late phase fluorescein angiogram showed hyperfluorescence caused by leakage from the choroid in the right (G) and left (H) eyes.
jkos-48-1716f2.tif
Figure 3.
B-scan ultrasonograms showed no thickening of the choroid and sclera in the right (A) and left (B) eye.
jkos-48-1716f3.tif
Figure 4.
The horizontal OCT image 7 weeks later showed that the RPE detachment appears slightly worse than on the previous exam in the left eye.
jkos-48-1716f4.tif
Figure 5.
(A) The horizontal OCT image 3 months later showed improvement of detachment of the neurosensory retina and RPE detachment in the left eye. Early (B) and late (D) phase fluorescein angiograms 3 months later remain unchanged in the right eye. Blockage of choroidal fluorescence in the early phase (C) and hyperfluorescence caused by leakage from the choroid in the late phase (E) in the left eye appear worse than on the previous examination.
jkos-48-1716f5.tif
Figure 6.
Fundus photograph 9 months later showed resolution of retinal infiltrates in the right (A) and left (B) eye. The horizontal OCT image 9 months later showed resolution of detachment of the neurosensory retina and RPE detachment in the right (C) and left (D) eye.
jkos-48-1716f6.tif
TOOLS
Similar articles