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Early treatment of idiopathic vasculitis, aneurysms and neuroretinitis (IRVAN). A case report
Tratamiento precoz de vasculitis idiopática, aneurismas y retinitis (IRVAN). A propósito de un caso
M. Garcia Vilaro
, F. Bassaganyas Vilarrassa, J.I. Vela Segarra, J.A. Buil Calvo
Corresponding author
Servicio de Oftalmología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain
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A case report" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "117" "paginaFinal" => "120" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M. Garcia Vilaro, F. Bassaganyas Vilarrassa, J.I. Vela Segarra, J.A. Buil Calvo" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Garcia Vilaro" "email" => array:1 [ 0 => "martagvilaro@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "F." "apellidos" => "Bassaganyas Vilarrassa" ] 2 => array:2 [ "nombre" => "J.I." "apellidos" => "Vela Segarra" ] 3 => array:2 [ "nombre" => "J.A." "apellidos" => "Buil Calvo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Oftalmología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tratamiento precoz de vasculitis idiopática, aneurismas y retinitis (IRVAN). A propósito de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 823 "Ancho" => 931 "Tamanyo" => 93950 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Retinograph of the left eye. Arterial aneurysms in posterior pole. Dense lipidic exudates in macular area following the pathway of large vessels.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">The IRVAN syndrome is a rare vascular disease described by Chang in 1995, characterized by idiopathic retinal vasculitis, posterior pole multiple arterial aneurysms and peripheral capillary occlusion.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It expresses in young and healthy individuals, preferably females. Visual prognosis is uncertain and even though some self-limited cases have been described with good visual acuity, peripheral ischemia and neovascularization are aggressive.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Clinic case</span><p id="par0010" class="elsevierStylePara elsevierViewall">Caucasian female, 55, without relevant pathological history, who visited the center in January 2008 due to diminished visual acuity in the left eye. Best corrected visual acuity was of 20/20 in the right eye and 20/60 in the left eye. Anterior segment exploration and tonometry did not show alterations. Ocular fundus examination revealed multiple large and medium-sized aneurysmatic dilatations in arteries and vasculitis in both eyes (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). The left eye exhibited lipidic exudation in the macular area (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Fluorescein angiography revealed large areas of peripheral ischemia without neovascularization and generalized vasculitis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Optic coherence tomography (OCT) showed macular lipidic deposits and diffuse macular edema.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Detailed systemic study was carried out with hemogram, glycemia, urine analysis, proteingram, ANCA, ANA, FR, VSG, ECA and cranial MR to discard associated pathology, which gave negative results. Relatives of the patient did not refer vascular malformation history.</p><p id="par0020" class="elsevierStylePara elsevierViewall">According to the new classification proposed by Samuel, the patient was diagnosed with IRVAN syndrome in stage 2.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was treated with confluent of peripheral panretinal photocoagulation in both eyes.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In subsequent follow-up visits, macular lipidic exudation in the left eye was diminished and the ocular fundus lesions had become stable (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>).</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">After 3 years follow-up, visual acuity was of 20/20 in the right eye and 20/40 in the left eye. The vascular alterations remained stable without progression of ischemia or neovascularization (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>).</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">An IRVAN syndrome case in initial stage, treated early prior to the development of neovascularization with peripheral panretinal photocoagulation which remains stable with a three-year follow-up period. After applying the treatment, lipidic exudation in the left eye posterior pole diminished and visual acuity improved (<a class="elsevierStyleCrossRef" href="#fig0025">Fig. 5</a>). Even though some authors have described aneurysm regression after treatment, the number and size of aneurysm-like dilatations did not exhibit variations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><elsevierMultimedia ident="fig0025"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">Samuel et al. defended the need of early ischemia treatment in order to control the progression of the disease.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The case presented herein involves favorably after treatment with peripheral panretinal photocoagulation in initial stages, thereby supporting said theory.</p><p id="par0050" class="elsevierStylePara elsevierViewall">In advanced stages of the disease (stages 3, 4 and 5) the visual prognosis is uncertain despite the treatment and there is high risk of developing complications secondary to retinal ischemia.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4,5</span></a> Due to the poor results obtained with standard treatments in these patients, some authors have utilized intravitreal anti-VEGF (ranibizumab and bevacizumab) prior to laser coagulation with good results.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflict of interests</span><p id="par0055" class="elsevierStylePara elsevierViewall">No conflict of interests has been declared by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres352612" "titulo" => array:3 [ 0 => "Abstract" 1 => "Case report" 2 => "Discussion" ] ] 1 => array:2 [ "identificador" => "xpalclavsec334076" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres352613" "titulo" => array:3 [ 0 => "Resumen" 1 => "Caso clínico" 2 => "Discusión" ] ] 3 => array:2 [ "identificador" => "xpalclavsec334075" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interests" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2011-01-25" "fechaAceptado" => "2012-07-12" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec334076" "palabras" => array:5 [ 0 => "Vasculitis" 1 => "Aneurysm" 2 => "Neuroretinitis" 3 => "Treatment" 4 => "Laser coagulation" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec334075" "palabras" => array:5 [ 0 => "Vasculitis" 1 => "Aneurismas" 2 => "Neuroretinitis" 3 => "Tratamiento" 4 => "Panfotocoagulación" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 55-year-old woman presented with retinal vasculitis, multiple aneurysms, macular exudation and widespread retinal nonperfusion and was diagnosed with IRVAN. She was treated with panretinal laser photocoagulation. After 3 years of follow-up visual acuity remains stable and there are no complications due to ischemic sequelae.</p> <span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">IRVAN syndrome with neovascularization can progress rapidly despite laser treatment. Panretinal laser photocoagulation has to be considered in the early stages as it is effective in stopping the progression of ischemia.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span class="elsevierStyleSectionTitle" id="sect0025">Caso clínico</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Se presenta el caso de una mujer de 55 años con vasculitis retiniana, aneurismas arteriales múltiples en el polo posterior, exudación macular e hipoperfusión periférica bilateral (IRVAN) tratada precozmente con panfotocoagulación periférica. Tras 3 años de seguimiento la agudeza visual permanece estable y no hay progresión de la isquemia.</p> <span class="elsevierStyleSectionTitle" id="sect0030">Discusión</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El síndrome de IRVAN con neovascularización es difícil de controlar y el riesgo de progresión a glaucoma neovascular, a pesar del tratamiento, es alto. La panfotocoagulación en estadios iniciales es eficaz para controlar la isquemia y debe realizarse lo antes posible.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Garcia Vilaro M, Bassaganyas Vilarrassa F, Vela Segarra JI, Buil Calvo JA. Tratamiento precoz de vasculitis idiopática, aneurismas y retinitis (IRVAN). A propósito de un caso. Arch Soc Esp Oftalmol. 2014;89:117–120.</p>" ] ] "multimedia" => array:5 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 780 "Ancho" => 930 "Tamanyo" => 75140 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Retinograph of the right eye, showing large diameter loop-shaped aneurysms and vascular sheathing.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 823 "Ancho" => 931 "Tamanyo" => 93950 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Retinograph of the left eye. Arterial aneurysms in posterior pole. Dense lipidic exudates in macular area following the pathway of large vessels.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1271 "Ancho" => 1447 "Tamanyo" => 153730 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Angiofluoresceingraph showing increased distal vascular patency.</p>" ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Fig. 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 593 "Ancho" => 1446 "Tamanyo" => 111495 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Control retinograph after 3 years. Diminished lipidic exudation in the left eye macular area and stability of vascular lesions. Confluent laser coagulation impacts can be observed.</p>" ] ] 4 => array:7 [ "identificador" => "fig0025" "etiqueta" => "Fig. 5" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr5.jpeg" "Alto" => 525 "Ancho" => 1446 "Tamanyo" => 148248 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">(A) Optic coherence tomography image of the macular area at diagnostic, showing intraretinal hyper-reflectiveness points corresponding to lipidic deposits and intraretinal edema. (B) Control optic coherence tomography 3 years later. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2023 March | 1 | 2 | 3 |
| 2018 February | 5 | 1 | 6 |
| 2018 January | 13 | 0 | 13 |
| 2017 December | 4 | 0 | 4 |
| 2017 November | 13 | 2 | 15 |
| 2017 October | 23 | 1 | 24 |
| 2017 September | 9 | 4 | 13 |
| 2017 August | 22 | 2 | 24 |
| 2017 July | 27 | 6 | 33 |
| 2017 June | 35 | 5 | 40 |
| 2017 May | 29 | 1 | 30 |
| 2017 April | 44 | 2 | 46 |
| 2017 March | 50 | 8 | 58 |
| 2017 February | 30 | 0 | 30 |
| 2017 January | 28 | 0 | 28 |
| 2016 December | 23 | 4 | 27 |
| 2016 November | 26 | 3 | 29 |
| 2016 October | 40 | 4 | 44 |
| 2016 September | 45 | 3 | 48 |
| 2016 August | 30 | 1 | 31 |
| 2016 July | 18 | 1 | 19 |
| 2016 June | 15 | 6 | 21 |
| 2016 May | 27 | 11 | 38 |
| 2016 April | 22 | 9 | 31 |
| 2016 March | 26 | 8 | 34 |
| 2016 February | 18 | 12 | 30 |
| 2016 January | 37 | 15 | 52 |
| 2015 December | 34 | 14 | 48 |
| 2015 November | 19 | 7 | 26 |
| 2015 October | 32 | 4 | 36 |
| 2015 September | 18 | 4 | 22 |
| 2015 August | 42 | 5 | 47 |
| 2015 July | 25 | 8 | 33 |
| 2015 June | 17 | 3 | 20 |
| 2015 May | 25 | 2 | 27 |
| 2015 April | 25 | 6 | 31 |
| 2015 March | 28 | 8 | 36 |
| 2015 February | 16 | 1 | 17 |
| 2015 January | 14 | 6 | 20 |
| 2014 December | 27 | 9 | 36 |
| 2014 November | 23 | 5 | 28 |
| 2014 October | 30 | 9 | 39 |
| 2014 September | 58 | 11 | 69 |
| 2014 August | 4 | 2 | 6 |
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