Treatment of Recalcitrant Idiopathic Orbital Inflammation (Chronic Orbital Myositis) With Infliximab JAMES A. GARRITY, MD, AUSTIN W. COLEMAN, DO, ERIC L. MATTESON, MD, MPH, ERIC R. EGGENBERGER, DO AND DAVID M. WAITZMAN, MD ● PURPOSE: To report results of treatment with a mono- clonal antibody (infliximab) directed against tumor ne- crosis factor  in seven patients with chronic and difficult-to-control idiopathic orbital inflammation (or- bital myositis). ● DESIGN: Observational case series. ● METHODS: Retrospective data were collected from seven patients who had idiopathic orbital inflammation and who were evaluated at three medical centers. All patients were treated with infliximab after the failure of traditional ther- apy, which included corticosteroids, radiotherapy, or anti- inflammatory chemotherapeutic agents. ● RESULTS: All seven patients had a favorable response to treatment with infliximab. One patient with Behçet disease required supplemental oral corticosteroids. Pain, swelling, and need for concomitant corticosteroids were the primary measures of treatment success. Symptoms of comorbid disease in four patients also improved (Crohn disease in two, Behçet disease in one, and psoriasis in one). There were no untoward effects of treatment after a mean fol- low-up of 15.7 months (range, 4 to 31 months). ● CONCLUSIONS: Treatment with infliximab appears to offer another therapeutic option in cases of recalcitrant or recurrent idiopathic orbital inflammation in which conventional treatment fails. (Am J Ophthalmol 2004; 138:925–930. © 2004 by Elsevier Inc. All rights re- served.) “ I DIOPATHIC ORBITAL INFLAMMATION” IS A NONSPE- cific term that refers to inflammation affecting various orbital structures. The inflammation may affect any orbital tissue, resulting in, for example, myositis, dacryoad- enitis, optic perineuritis, or scleritis, or all of the various orbital tissues. The condition may be acute or chronic. 1–6 It typically responds to high doses of corticosteroids, but this therapy subjects patients to the well-known hazards of chronic corticosteroid use, and the response even to high doses can be unsatisfactory. 7,8 Inflammation may recur when the corticosteroid dosage is decreased. These side effects often make it necessary to consider alternative anti-inflammatory medications for some patients. Metho- trexate, cyclophosphamide, other antineoplastic agents, and nonsteroidal anti-inflammatory drugs have been used to decrease or alleviate the dosage requirement of cortico- steroid therapy. 9 Radiotherapy has also been used as a treatment alternative; the risks include dry eye, cataract formation, retinopathy, optic neuropathy, and secondary malignancy. 9,10 DESIGN THIS STUDY WAS A RETROSPECTIVE, OBSERVATIONAL CASE series. METHODS THERAPY WITH TUMOR NECROSIS FACTOR (TNF)  INHIBI- tors, such as infliximab, is efficacious for immune-mediated inflammatory conditions, including rheumatoid arthritis and Crohn disease, 11–15 but their use for inflammatory orbital pseudotumor is not well described. We describe seven cases of chronic or recurrent idiopathic orbital inflammation (orbital myositis), which responded well to infliximab administration. A summary of the cases is presented in Table 1. Comorbid conditions were present in most patients. All patients under- went clinical and laboratory investigation for other systemic inflammatory/autoimmune conditions, including sarcoidosis, Graves disease, systemic lupus erythematosus, idiopathic in- flammatory myositis, and other forms of vasculitis, which were not detected other than as described. Accepted for publication June 22, 2004. From the Department of Ophthalmology (J.A.G.) and the Division of Rheumatology (E.L.M.), Mayo Clinic, Rochester, Minnesota; the Depart- ments of Ophthalmology and Neurology (A.W.C., E.R.E.), Michigan State University, East Lansing, Michigan USA; and the University of Connecticut Health Center (D.M.W.), Farmington, Connecticut. Inquiries to James A. Garrity, MD, Department of Ophthalmology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905; fax: 507-284- 4612; e-mail: garrity.james@mayo.edu © 2004 BY ELSEVIER INC.ALL RIGHTS RESERVED. 0002-9394/04/$30.00 925 doi:10.1016/j.ajo.2004.06.077