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      Rapid Regression of Exudative Maculopathy in Idiopathic Retinitis, Vasculitis, Aneurysms and Neuroretinitis Syndrome after Intravitreal Ranibizumab

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          Abstract

          The idiopathic retinitis, vasculitis, aneurysms and neuroretinitis syndrome is a rare retinal vascular disorder characterized by multiple leaking aneurysmal dilations, retinal vasculitis, neuroretinitis and peripheral vascular ischemia. Visual loss mainly occurs due to the development of retinal neovascularization and/or exudative maculopathy. Although the treatment of choice has not yet been established, retinal photocoagulation seems to be the best option to control the disease and to prevent its progression. Herein, we report a case of idiopathic retinitis, vasculitis, aneurysms and neuroretinitis syndrome with both retinal neovascularization and macular exudation successfully managed with intravitreal ranibizumab (Lucentis ®) as adjunctive therapy to retinal photocoagulation.

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          Subgroup analysis of the MARINA study of ranibizumab in neovascular age-related macular degeneration.

          , Robert Bhisitkul, Vidya Acharya … (2007)
          An examination of clinically relevant subgroups of patients in the MARINA study of ranibizumab in treatment of minimally classic or occult with no classic choroidal neovascularization (CNV) secondary to age-related macular degeneration (AMD) was done. Objectives were to determine the effectiveness of ranibizumab across subgroups, compare the effectiveness of ranibizumab with that of sham injection within subgroups, and evaluate the relationship between selected baseline characteristics and visual acuity (VA) outcomes. Retrospective subgroup analyses of 24-month data from the MARINA study. Seven hundred sixteen patients were randomly assigned to 0.3 mg ranibizumab (n = 238), 0.5 mg ranibizumab (n = 240), or sham treatment (n = 238). Efficacy outcomes were compared across subgroups based on patients' gender, age, baseline VA score, baseline CNV lesion size, CNV lesion type, and duration of neovascular AMD using univariate analyses. Multivariate analyses were performed on the change from baseline to 24 months in VA score to assess further the correlation between baseline characteristics and VA outcome. Proportion of patients losing or =15 letters from baseline, and mean VA score change from baseline. For each of the 3 VA end points, all subgroups of ranibizumab-treated patients did better on average than the sham-treated patients. Increasing age, larger CNV lesion size at baseline, and a higher baseline VA score were all associated with greater loss of letters in the sham group or less gain of letters in the ranibizumab groups. However, the net benefit of ranibizumab versus sham treatment was greater in patients who scored higher than in those who scored lower in baseline VA. This subgroup analysis of 24-month data from the MARINA study indicates that ranibizumab treatment was associated with an average increase from baseline VA in all subgroups evaluated, and that ranibizumab treatment was superior to sham treatment across all subgroups. The most important predictors of VA outcomes were, in decreasing order of importance, baseline VA score, CNV lesion size, and age.
          Article 0 comments Cited 108 times – based on 0 reviews     Review now
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            Idiopathic retinitis, vasculitis, aneurysms, and neuroretinitis (IRVAN): new observations and a proposed staging system.

            R Equi, William F. Mieler, Alastair D Hay … (2007)
            To review the clinical features, disease progression, and effects of treatment on idiopathic retinitis, vasculitis, aneurysms, and neuroretinitis (IRVAN). Retrospective interventional case series. Ten patients with IRVAN originally reported in 1995 and 12 additional patients identified since the original series. Patients in the series had testing that may have included fluorescein angiography, indocyanine green angiography, and systemic evaluation. Treatments included panretinal laser photocoagulation, cryotherapy, vitrectomy surgery, and injection of periocular or intravitreal steroids. Initial visual acuity (VA), initial stage at diagnosis, clinical course, surgical intervention, final VA, and complications of disease. A total of 44 eyes of 22 patients were studied; 9 eyes had reached stage 1 or 2 disease at last follow-up, 17 had reached stage 3, and 12 had reached stage 4 or 5. At the time of last follow-up, 14 eyes had maintained 20/20 vision, 15 had between 20/40 and 20/200 vision, and 9 had 20/300 vision or worse. Later stages of retinal ischemia are associated with worse VA. Thirty-two of 38 followed eyes were treated. Twenty-five were treated initially with panretinal laser photocoagulation. The clinical course of each eye after initiation of panretinal laser photocoagulation was evaluated with respect to the final VA and stage of ischemic retinopathy at the initiation of treatment. Panretinal laser photocoagulation was initiated in 3 eyes at stage 2, 16 at stage 3, 5 at stage 4, and 1 at stage 5. Seven eyes underwent grid laser retinal photocoagulation of the macula for macular edema. Idiopathic retinitis, vasculitis, aneurysms, and neuroretinitis is an isolated retinal vascular disease that can progress rapidly to severe vision loss due to ischemic sequelae despite treatment with panretinal laser photocoagulation. Based on our review of the largest cohort of IRVAN patients, early panretinal laser photocoagulation should be considered when angiographic evidence of widespread retinal nonperfusion is present, and before (or shortly after) the development of neovascularization. A functional staging system is proposed to improve treatment paradigms.
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              Idiopathic retinal vasculitis, aneurysms, and neuro-retinitis. Retinal Vasculitis Study.

              W Mieler, G Aylward, J Gass … (1995)
              The authors describe the clinical feature of ten patients with a new syndrome characterized by the presence of retinal vasculitis, multiple macroaneurysms, neuro-retinitis, and peripheral capillary nonperfusion. The authors evaluated ten patients identified to have clinical features compatible with the syndrome of idiopathic retinal vasculitis, aneurysms and neuroretinits (IRVAN). Clinical examination findings, sequential funds photographs (when available), fluorescein angiograms, systemic investigations, response to therapy, and visual outcomes were reviewed. Seven eyes of four patients sustained a marked decrease in visual acuity of 20/200 or worse. Visual loss was due to a combination of an exudative maculopathy and sequelae of retinal ischemia. Capillary nonperfusion was seen in all ten patients and was severe enough to warrant panretinal laser photocoagulation in six patients. Systemic investigations were uniformly noncontributory. Oral prednisone appears to have little beneficial effects on patients with this disorder. Patients with IRVAN have characteristic retinal features that readily identify this syndrome. An increased awareness of this rare syndrome may help to identify sight-threatening complications at an earlier stage. The authors caution against extensive medical investigations.
              Article 0 comments Cited 18 times – based on 0 reviews     Review now
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                Author and article information

                Journal
                Journal ID (nlm-ta): Case Report Ophthalmol
                Journal ID (iso-abbrev): Case Report Ophthalmol
                Journal ID (publisher-id): COP
                Title: Case Reports in Ophthalmology
                Publisher: S. Karger AG (Allschwilerstrasse 10, P.O. Box · Postfach · Case postale, CH–4009, Basel, Switzerland · Schweiz · Suisse, Phone: +41 61 306 11 11, Fax: +41 61 306 12 34, karger@karger.ch )
                ISSN (Electronic): 1663-2699
                Publication date Collection: May-Aug 2012
                Publication date (Electronic): 15 August 2012
                Publication date PMC-release: 15 August 2012
                Volume: 3
                Issue: 2
                Pages: 251-257
                Affiliations
                [1] aDepartment of Ophthalmology, University and Polytechnic Hospital La Fe, Alzira, Spain
                [2] bFaculty of Medicine, Valencia University, Valencia, Alzira, Spain
                [3] cDepartment of Ophthalmology, La Ribera University Hospital, Alzira, Spain
                Author notes
                *Cristina Marín-Lambíes Querol, 7-14, ES-46002 Valencia (Spain), Tel. +34 657 887 201, E-Mail crismarinlambies@ 123456hotmail.com
                Article
                Publisher ID: cop-0003-0251
                DOI: 10.1159/000341821
                PMC ID: 3433025
                PubMed ID: 22949913
                SO-VID: a7da9d90-353c-4b74-951a-7ba8c6897ea1
                Copyright © Copyright © 2012 by S. Karger AG, Basel
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-Noncommercial-No-Derivative-Works License ( http://creativecommons.org/licenses/by-nc-nd/3.0/). Users may download, print and share this work on the Internet for noncommercial purposes only, provided the original work is properly cited, and a link to the original work on http://www.karger.com and the terms of this license are included in any shared versions.

                History
                Page count
                Figures: 2, References: 15, Pages: 7
                Categories
                Subject: Published online: August, 2012

                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: idiopathic retinitis, vasculitis, aneurysms and neuroretinitis syndrome,exudative maculopathy,ranibizumab
                Data availability:
                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: idiopathic retinitis, vasculitis, aneurysms and neuroretinitis syndrome, exudative maculopathy, ranibizumab

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