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      Adalimumab for Treatment of Idiopathic Frosted Branch Angiitis: A Case Report

      case-report

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          Abstract

          Purpose

          To report a case of recurrent idiopathic frosted branch angiitis (FBA) successfully treated with adalimumab.

          Case Report

          A 14-year-old otherwise healthy boy was referred to the uveitis clinic for bilateral panuveitis with diffuse retinal vascular sheathing and severe macular edema. Extensive work-up including aqueous sampling for detection of viral causes was inconclusive. Two years ago, a similar episode had been treated with oral prednisolone, however it was complicated by adverse psychiatric effects. The progressive course of the condition mandated considering other therapeutic measures; Adalimumab was chosen based on its purported efficacy for treatment of childhood uveitis and a favorable safety profile. The patient responded dramatically to a single subcutaneous injection of adalimumab without any side effect during and after injection. The therapeutic effect was rapid and relatively long-lasting.

          Conclusion

          To the best of our knowledge, this is the first case of idiopathic FBA treated successfully with adalimumab without adjunctive steroid therapy.

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          Most cited references6

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          Frosted branch angiitis: a review.

          Adam Walker, Andrea N. Jones, A Iguchi (2004)
          The purpose of this study is to present the first report of a case of primary frosted branch angiitis from the UK and to review the characteristics of this rare disease. Primary frosted branch angiitis causes characteristic florid translucent retinal perivascular sheathing of both arterioles and venules in association with variable uveitis, retinal oedema and visual loss, normally with good recovery. A total of 57 cases have been reported in the world literature. Atypical, typically focal frosted branch angiitis may also occur secondary to other causes of intraocular inflammation, especially cytomegalovirus retinitis. Primary frosted branch angiitis has a characteristic presentation but a variable course, typically affecting children or young adults. The disease is likely to represent a common immune pathway in response to multiple infective agents. The optimal treatment is unclear.
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            Frosted branch angiitis: clinical syndrome or clinical sign?

            R Kleiner (1996)
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              Long-term efficacy of adalimumab in the treatment of uveitis associated with juvenile idiopathic arthritis

              Kaisu Kotaniemi, Hanna Säilä, Hannu Kautiainen (2011)
              Background The purpose of this study was to investigate the long-term effects of adalimumab, a tumor necrosis factor alpha antagonist, in the treatment of uveitis associated with juvenile idiopathic arthritis. Methods Adalimumab was initiated in 94 patients with juvenile idiopathic arthritis to treat active arthritis and/or active associated uveitis. In 18 patients, therapy was discontinued after a short period because of inefficacy or side effects. The activity of uveitis (using Standardized Uveitis Nomenclature [SUN] criteria and clinical examination) and arthritis (number of swollen or active joints) was evaluated at the start and at end of the study. Results At the end of the study, uveitis was under good clinical control in two thirds of 54 patients (31% did not need any local treatment and 35% used only 1–2 corticosteroid drops a day), and one third had active uveitis (at least three corticosteroid drops a day). According to SUN criteria, adalimumab treatment for uveitis showed improved activity (a two-fold decrease in uveitis activity) in 28% of patients, with a moderate response in 16 patients, no change in a further 16 patients, and worsening activity (a two-fold increase in uveitis activity) in 13% of patients. The overall proportion of patients with active arthritis decreased. At the beginning of the study, 69% of patients with uveitis had more than two active joints, and at the end of the study only 27% had active joint disease. In 27 patients with juvenile idiopathic arthritis without uveitis on adalimumab, the number of active joints decreased from 93% to 59%. Systemic corticosteroid treatment could be stopped in 22% of patients with uveitis and in 11% of those without uveitis. Most of the patients had received methotrexate, other immunosuppressive therapy, or other biological drugs before initiating adalimumab. Conclusion Adalimumab is a valuable option in the treatment of uveitis associated with active juvenile idiopathic arthritis.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Ophthalmic Vis Res
                Journal ID (iso-abbrev): J Ophthalmic Vis Res
                Journal ID (publisher-id): JOVR
                Title: Journal of Ophthalmic & Vision Research
                Publisher: Ophthalmic Research Center
                ISSN (Print): 2008-2010
                ISSN (Electronic): 2008-322X
                Publication date (Print): October 2013
                Volume: 8
                Issue: 4
                Pages: 372-375
                Affiliations
                [1 ]Noor Ophthalmic Research Center, Noor Eye Hospital, Tehran, Iran
                [2 ]Eye Research Center, Rassoul Akram Hospital, Iran University of Medical Sciences, Tehran, Iran
                [3 ]Ophthalmic Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran
                Author notes
                Correspondence to: Alireza Hedayatfar, MD. Noor Eye Hospital, #96 Esfandiar Blvd., Vali’asr Ave., Tehran, 196865311, Iran; Tel: +98 21 82400, Fax: +98 21 8865 0501; e-mail: ahedaiatfar@ 123456noorvision.com
                Article
                Publisher ID: JOVR-08-372
                PMC ID: 3957044
                PubMed ID: 24653825
                SO-VID: 5382dc98-230c-479b-ace5-b5322649f78a
                Copyright © © 2013 Ophthalmic Research Center, Shahid Beheshti University of Medical Sciences
                License:

                This work is licensed under a Creative Commons Attribution-NonCommercial 3.0 Unported License which allows users to read, copy, distribute and make derivative works for non-commercial purposes from the material, as long as the author of the original work is cited properly.

                History
                Date received : 16 June 2012
                Date accepted : 07 January 2013
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: adalimumab,anti-tumor necrosis factor,frosted branch angiitis,uveitis
                Data availability:
                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: adalimumab, anti-tumor necrosis factor, frosted branch angiitis, uveitis

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