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Park, Woo, and Kim: Three Cases of Rituximab Treatment for Orbital Inflammatory Disease
Case Report
Journal of the Korean Ophthalmological Society

Journal of the Korean Ophthalmological Society 2012; 53(5): 721-727.

Published online: 16 May 2012

DOI: https://doi.org/10.3341/jkos.2012.53.5.721

Three Cases of Rituximab Treatment for Orbital Inflammatory Disease

Byung-Chun Park, MD1, Kyung-In Woo, MD, PhD2, Yoon-Duck Kim, MD, PhD2

1Department of Ophthalmology, Dongkang Medical Center, Ulsan, Korea.

2Department of Ophthalmology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.

Address reprint requests to Yoon-Duck Kim, MD, PhD. Department of Ophthalmology, Sungkyunkwan University Samsung Medical Center, #81 Irwon-ro, Gangnam-gu, Seoul 135-710, Korea. Tel: 82-2-3410-3570, Fax: 82-2-3410-0074, ydkimoph@skku.edu

Received 4 May 2011       Accepted 24 March 2012

Copyright © 2012 The Korean Ophthalmological Society

Abstract

Purpose

To report three cases of rituximab treatment in refractory orbital inflammatory disease.

Case summary

Rituximab treatment was performed in three patients (thyroid-associated ophthalmopathy, orbital myositis, and idiopathic sclerosing inflammation) unresponsive to corticosteroid and radiotherapy. One cycle consisted of intravenous infusion of 1000 mg of rituximab, twice at a two-week interval. The patient with thyroid-associated ophthalmopathy exhibited clinical improvement in visual acuity, color vision, visual evoked potential, and visual field after one cycle of rituximab treatment. The patient with orbital myositis improved showed diplopia with a decrease in the size of the extraocular muscle after two cycles of treatment. The patient with idiopathic sclerosing inflammation showed improvements in visual acuity and visual field defect after one cycle of therapy. There were no severe infections or hematologic adverse effects including neutropenia, or decreased immunoglobulin during the follow-up period.

Conclusions

Treatment with rituximab appears to offer another therapeutic option in patients unresponsive to corticosteroid or radiotherapy.

Keywords: Inflammation, Orbit, Rituximab, Thyoid

Figures and Tables

Figure 1
Case 1. (A, C) Both eyes show conjunctival injection and proptosis. (B, D) Conjunctival injection in both eyes have markedly decreased after rituximab infusion. (E, G) Axial (E) and coronal (G) CT scans show marked fusiform enlargement of extraocular muscles in both sides. The coronal section shows compression of optic nerve in the orbital apex. (F, H) After rituximab infusion, apical crowding and stretching of optic nerve in the CT scan have improved. (I, J) Change in visual field before (I) and after (J) rituximab infusion.
jkos-53-721-g001
Figure 2
Case 2. (A, B) Proptosis and upper lid swelling of the left eye before rituximab infusion (A) decreased after therapy (B). (C, E) Coronal CT scans show enlargement of the left superior oblique and medial rectus before rituximab treatment. (D, F) After rituximab treatment, muscle enlargement markedly decreased.
jkos-53-721-g002
Figure 3
Case 3. (A, C) CT scans show infiltrative lobulated homogeneous mass in the left intraconal space. The optic nerve is involved by the orbital mass and displaced in the inferolateral direction. (B, D) After Rituximab infusion, orbital mass decreased and optic nerve infiltration improved.
jkos-53-721-g003

References

1. Lutt JR, Lim LL, Phal PM, Rosenbaum JT. Orbital inflammatory disease. Semin Arthritis Rheum. 2008. 37:207–222.
2. Tsokos GC. B cells, be gone--B-cell depletion in the treatment of rheumatoid arthritis. N Engl J Med. 2004. 350:2546–2548.
3. Pescovitz MD. Rituximab, an anti-cd20 monoclonal antibody: history and mechanism of action. Am J Transplant. 2006. 6(5 Pt 1):859–866.
4. Smolen JS, Keystone EC, Emery P, et al. Consensus statement on the use of rituximab in patients with rheumatoid arthritis. Ann Rheum Dis. 2007. 66:143–150.
5. El Fassi D, Nielsen CH, Hasselbalch HC, Hegedus L. The rationale for B lymphocyte depletion in Graves' disease. Monoclonal anti-CD20 antibody therapy as a novel treatment option. Eur J Endocrinol. 2006. 154:623–632.
6. Kurz PA, Suhler EB, Choi D, Rosenbaum JT. Rituximab for treatment of ocular inflammatory disease: a series of four cases. Br J Ophthalmol. 2009. 93:546–548.
7. Harris GJ. Idiopathic orbital inflammation: a pathogenetic construct and treatment strategy: The 2005 ASOPRS Foundation Lecture. Ophthal Plast Reconstr Surg. 2006. 22:79–86.
8. Yuen SJ, Rubin PA. Idiopathic orbital inflammation: distribution, clinical features, and treatment outcome. Arch Ophthalmol. 2003. 121:491–499.
9. Rubin PA, Foster CS. Etiology and management of idiopathic orbital inflammation. Am J Ophthalmol. 2004. 138:1041–1043.
10. Hougardy DM, Peterson GM, Bleasel MD, Randall CT. Is enough attention being given to the adverse effects of corticosteroid therapy? J Clin Pharm Ther. 2000. 25:227–234.
11. Silkiss RZ, Reier A, Coleman M, Lauer SA. Rituximab for thyroid eye disease. Ophthal Plast Reconstr Surg. 2010. 26:310–314.
12. Kazlow Stern D, Tripp JM, Ho VC, Lebwohl M. The use of systemic immune moderators in dermatology: an update. Dermatol Clin. 2005. 23:259–300.
13. Nash PT, Florin TH. Tumour necrosis factor inhibitors. Med J Aust. 2005. 183:205–208.
14. Ho VH, Chevez-Barrios P, Jorgensen JL, et al. Receptor expression in orbital inflammatory syndromes and implications for targeted therapy. Tissue Antigens. 2007. 70:105–109.
15. Nielsen CH, El Fassi D, Hasselbalch HC, et al. B-cell depletion with rituximab in the treatment of autoimmune diseases. Graves' ophthalmopathy the latest addition to an expanding family. Expert Opin Biol Ther. 2007. 7:1061–1078.
16. Takemura S, Klimiuk PA, Braun A, et al. T cell activation in rheumatoid synovium is B cell dependent. J Immunol. 2001. 167:4710–4718.
17. Vos K, Thurlings RM, Wijbrandts CA, et al. Early effects of rituximab on the synovial cell infiltrate in patients with rheumatoid arthritis. Arthritis Rheum. 2007. 56:772–778.
18. Hasselbalch HC. B-cell depletion with rituximab-a targeted therapy for Graves' disease and autoimmune thyroiditis. Immunol Lett. 2003. 88:85–86.
19. Salvi M, Vannucchi G, Campi I, et al. Efficacy of rituximab treatment for thyroid-associated ophthalmopathy as a result of intraorbital B-cell depletion in one patient unresponsive to steroid immunosuppression. Eur J Endocrinol. 2006. 154:511–517.
20. Salvi M, Vannucchi G, Campi I, et al. Treatment of Graves' disease and associated ophthalmopathy with the anti-CD20 monoclonal antibody rituximab: an open study. Eur J Endocrinol. 2007. 156:33–40.
21. Khanna D, Chong KK, Afifiyan NF, et al. Rituximab treatment of patients with severe, corticosteroid-resistant thyroid-associated ophthalmopathy. Ophthalmology. 2010. 117:133–139.e2.
22. El Fassi D, Banga JP, Gilbert JA, et al. Treatment of Graves' disease with rituximab specifically reduces the production of thyroid stimulating autoantibodies. Clin Immunol. 2009. 130:252–258.
23. Schafranski MD. Idiopathic orbital inflammatory disease successfully treated with rituximab. Clin Rheumatol. 2009. 28:225–226.
24. Smolen JS, Keystone EC, Emery P, et al. Consensus statement on the use of rituximab in patients with rheumatoid arthritis. Ann Rheum Dis. 2007. 66:143–150.
25. Emery P, Fleischmann R, Filipowicz-Sosnowska A, et al. The efficacy and safety of rituximab in patients with active rheumatoid arthritis despite methotrexate treatment: results of a phase IIB randomized, double-blind, placebo-controlled, dose-ranging trial. Arthritis Rheum. 2006. 54:1390–1400.
26. Edwards JC, Szczepanski L, Szechinski J, et al. Efficacy of B-cell-targeted therapy with rituximab in patients with rheumatoid arthritis. N Engl J Med. 2004. 350:2572–2581.
27. Dunleavy K, Hakim F, Kim HK, et al. B-cell recovery following rituximab-based therapy is associated with perturbations in stromal derived factor-1 and granulocyte homeostasis. Blood. 2005. 106:795–802.
28. Edwards JC, Leandro MJ, Cambridge G. B lymphocyte depletion therapy with rituximab in rheumatoid arthritis. Rheum Dis Clin North Am. 2004. 30:393–403. viii
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