POSTSURGICAL SYMPATHETIC OPHTHALMIA REFRACTORY TO TREATMENT: THE RESPONSE TO INFLIXIMAB Emilio Dorronzoro Ramı´rez, PhD,* Juan Jacobo Gonza´lez Guijarro, MD,* Rosario Garcı´a Vicun˜a, MD†

Purpose: A clinical case of a postoperative sympathetic ophthalmia is presented and its response to the monoclonal antibody infliximab. Patients: A 61-year-old woman. Results: The treatment with prednisone, cyclosporine, azathioprine, and mycophenolate mofetil did not completely control the loss of visual acuity caused by the disease. Monthly intravenous infusions of infliximab (5 mg/kg) significantly improved the condition of the patient, allowing the cyclosporine and azathioprine to be withdrawn and the prednisone to be reduced to 10 mg/d. Conclusion: The use of the immunosuppressive drug infliximab in combination with prednisone improved the prognosis of a patient suffering from sympathetic ophthalmia. The reported effectiveness of infliximab in the treatment of T lymphocyte–mediated diseases suggests that it could be used earlier in cases of sympathetic ophthalmia when treatment with corticosteroids must be shortened. RETINAL CASES & BRIEF REPORTS 6:169–171, 2012

From the *Uveitis Department; and †Rheumatology Department, Hospital Universitario de La Princesa, Madrid, Spain.

Case Report We present the case of a 61-year-old woman who was seen in the Uveitis Department in March 2004 complaining of loss of visual acuity in her left eye. The most relevant factors in her medical history were left eye cataract surgery and phthisis in the right eye caused by long-term retinal detachment. The examination revealed a retinal detachment in the left eye. Scleral buckling and vitrectomy were successfully performed. Two months later, visual acuity decreased to 20/50 and multiple choroidal infiltrates were observed in the fundus (Figure 1). Late leakage from the areas of choroidal infiltrates and infrapapillary periphlebitis were also visible on fluorescein angiography (Figure 2). Based on these findings, the patient was diagnosed with sympathetic ophthalmia. Treatment with prednisone was prescribed, with a starting dose of 100 mg/d, being gradually decreased over a 3-month period. Visual acuity increased from 20/50 to 20/20, with noticeable improvement in lesion activity in the fundus seen on fluorescein angiography. At 20 mg/d, the inflammation resumed and visual acuity decreased to 20/ 30. The patient refused to have the phthisical eye removed or have an intravitreal injection of triamcinolone because of a risk of retinal detachment, so cyclosporine 5 mgkg21d21 was added. However, after 1 month on this regimen, the patient developed renal toxicity and the dose was therefore reduced to 75 mg/d and azathioprine 1.5 mgkg21d21 was added. The treatment did not completely control the disease and visual acuity decreased to 20/50. In March 2005, we added mycophenolate mofetil 1 g/d, which resulted in an improvement

S

ympathetic ophthalmia is a bilateral granulomatous panuveitis in both eyes caused by penetrating trauma or ocular surgery in one eye. The incidence is higher after nonsurgical trauma (0.2%) and is more frequent during surgery after pars plana vitrectomy (0.01%) or multiple surgical procedures (0.06%).1 Patients show a positive proliferative response with a predominance of T lymphocytes in uveal preparations.2 Infliximab is a chimeric (murine–human) monoclonal antibody against tumor necrosis factor alfa, a cytokine that plays an essential role in the induction and maintenance of the immune response through the activation of T cells and macrophages.3 Presented in poster format at the IOIS Meeting, Paris, France, June 9, 2007. The authors have no commercial interest in the publication of the article. Reprint requests: Emilio Dorronzoro Ramı´rez, PhD, Uveitis Department, Hospital Universitario de La Princesa, 28028 Madrid, Spain; e-mail: [email protected]

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Fig. 1. Multiple choroidal infiltrates with no vitritis were observed in the fundus.

in visual acuity to 20/25. The condition was controlled until October 2005, at which time there was a recurrence. Mycophenolate mofetil was withdrawn and monthly intravenous infusions of infliximab 5 mg/kg were started (Figure 3). The patient had less active infiltrates, and in January 2006, the cyclosporine was withdrawn and the dose of prednisone was reduced to 10 mg/d, maintaining the same doses of azathioprine and infliximab. By November 2006, the patient had improved (Figure 4), with a visual acuity of 20/20, and azathioprine was withdrawn. In January 2007, an attempt was made to reduce the dose of prednisone from 10 mg/d and increase the intervals between infliximab infusions, but the patient relapsed.

Fig. 3. October 2005. To treat this reactivation, mycophenolate mofetil was withdrawn and treatment with infliximab was started.

patients suffering from sympathetic ophthalmia, but long-term monitoring is necessary because of possible relapses and adverse side effects.1 No published cases (MEDLINE) were found on the use of infliximab in the treatment of sympathetic ophthalmia. Given the effectiveness of this drug for controlling T lymphocyte–mediated diseases, it could be used earlier in the treatment of sympathetic ophthalmia when doses of corticosteroids need to be reduced. Acknowledgments

Discussion We thank translator Fernando Expo´sito Ramı´rez. The use of corticosteroids combined with immunosuppressive drugs has improved the prognosis of

Key words: sympathetic ophthalmia, infliximab, immunosuppressive drugs, prognosis improvement.

Fig. 2. Hypofluorescence of the choroidal infiltrates with late leakage and infrapapillary periphlebitis could also be seen on fluorescein angiography.

Fig. 4. In November 2006, the patient was better with almost no active infiltrates and no periphlebitis.

POSTSURGICAL SYMPATHETIC OPHTHALMIA

References 1. Power WJ. Sympathetic ophthalmia. In: Foster CS, Vitale AT, eds. Diagnosis and Treatment of Uveitis. Philadelphia, PA: W. Saunders Company; 2002:742–747.

171 2. Romayne S. Young S, Lightman S. Immunopathology of the noninfectious posterior and intermediate uveitides. Surv Ophthalmol 2001;46:209–233. 3. Valesini G, Iannuccelli C, Marocchi E, et al. Biological and clinical effects of anti-TNFa treatment. Autoimmun Rev 2007;7:35–41.

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