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Congenital retinal macrovessel associated with retinal peripheral telangiectasia and retinal ischaemia
Macrovaso retiniano asociado a telangiectasias periféricas retinianas e isquemia retiniana
A. Medina-Tapia, F.E. Molina-Sócola, L. Llerena-Manzorro
, F. López-Herrero, M. Castilla-Martino, A. Martínez-Borrego, J.L. Sánchez-Vicente
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Servicio de Oftalmología, Hospital Universitario Virgen del Rocío, Sevilla, Spain
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"apellidos" => "Flores-Suárez" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0365669116302192" "doi" => "10.1016/j.oftal.2016.10.016" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0365669116302192?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579417300671?idApp=UINPBA00004N" "url" => "/21735794/0000009200000007/v1_201706250033/S2173579417300671/v1_201706250033/en/main.assets" ] "en" => array:20 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Congenital retinal macrovessel associated with retinal peripheral telangiectasia and retinal ischaemia" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "338" "paginaFinal" => "342" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "A. Medina-Tapia, F.E. Molina-Sócola, L. Llerena-Manzorro, F. López-Herrero, M. Castilla-Martino, A. Martínez-Borrego, J.L. Sánchez-Vicente" "autores" => array:7 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Medina-Tapia" ] 1 => array:2 [ "nombre" => "F.E." "apellidos" => "Molina-Sócola" ] 2 => array:4 [ "nombre" => "L." "apellidos" => "Llerena-Manzorro" "email" => array:1 [ 0 => "laurall_1988@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 3 => array:2 [ "nombre" => "F." "apellidos" => "López-Herrero" ] 4 => array:2 [ "nombre" => "M." "apellidos" => "Castilla-Martino" ] 5 => array:2 [ "nombre" => "A." "apellidos" => "Martínez-Borrego" ] 6 => array:2 [ "nombre" => "J.L." "apellidos" => "Sánchez-Vicente" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario Virgen del Rocío, Sevilla, 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" => "Macrovaso retiniano asociado a telangiectasias periféricas retinianas e isquemia retiniana" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1575 "Ancho" => 1500 "Tamanyo" => 279544 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">LE angiofluorescence, showing increased telangiectasiae associated to ischaemia areas.</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 term “congenital retinal macrovessel” (CRM) was first used by Brown in 1982 to describe an aberrant retinal vessel, generally a vein, that passes through the central macular irrigating or draining areas located above and below the horizontal raphe. This anomaly is usually unilateral and rarely affects visual acuity (VA).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> CRM is a rare expression which is not usually associated to visual defects except when vascular anomalies compromise foveal circulation. CRM are generally venous although on rare occasions they could also involve an artery and vein, and even more rarely could be arterial.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Normally, CRM is an isolated finding although it has been described in association to other retinal alterations such as macular preretinal hemorrhages, perifoveal microvascular alterations,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> central serous choroidopathy,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> arterial macroaneurysms,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> retinopathy of prematurity,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> vascular occlusion, cystic macular edema and vascular malformations of the central nervous system.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> However, to date the association between a CRM and the presence of peripheral retinal telangiectasiae has not been described.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The case of a young patient with a unilateral congenital retinal macrovein associated to peripheral telangiectasiae and retinal ischaemia areas is described.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Clinic case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">Female, 16, who consulted due to history of headaches and blurred vision. VA in the right eye (RE) was 1.0 and the left eye (LE) 1.0. Anterior pole biomicroscopy and intraocular pressure were normal. RE ocular fundus was normal. An asymmetry between the cup of both papilla was observed. The most striking finding was the presence in the LE of a large size, dilated and tortuous inferior temporal vein that crossed the horizontal raphe. Fluorescein angiography (FAG) revealed thickened walls and increased diameter of the inferior temporal vein that crossed the horizontal raphe in the temporal area to the macula. The vessel exhibited early filling with delayed emptying, without diffusion. The temporal mean periphery exhibited telangiectasiae and non-exudative arteriovenous anastomosis associated to areas of limited ischaemia. No signs of macular loss in late times were observed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Angiographic optical coherence tomography (angio-OCT) (DRI OCT Triton [plus], Topcon Medical Systems, Inc., Europe) did not show alterations excepting the presence of said macrovessel (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Association with systemic diseases with other vascular malformations such as the Bonnet–Dechaume–Blanc or the Wyburn–Mason syndromes, as well as the absence of diseases that cause a secondary telangiectasia were discarded with angio-TAC. Accordingly, it was decided to maintain observation of the case. At follow-up month 11, the patient maintained VA of 1.0 in both eyes. FAG revealed increased telangiectasiae and ischaemia in the temporal quadrant (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Considering the situation, laser photocoagulation of said ischemic areas was performed (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>). Three months after said treatment, VA remains stable without telangiectasiae growth or appearance of new ischemic areas.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">CRM occurred during the differentiation and canalization of the mesenchymal cell cords that invade the retinal nerve fibers in gestation week 15–16. The development of the retina and vascular components are independent of each other and for this reason VA problems are not usually encountered except when these changes involve foveal microcirculation.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">CRM has been associated to diminished VA as the result of macular hemorrhages, foveolar cysts, serous macular detachments and presence of anomalous vessels in the fovea. CRA has also been described together with capillary perfusion alterations, arteriovenous communication and increased afoveal avascular area.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> However, after reviewing the published literature, the authors have not found association with peripheral retinal telangiectasia as in the case discussed herein.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Retinal telangiectasiae are vascular alterations characterized by the loss of endothelial cells and pericytes. These changes give rise to the loss of the blood–retina barrier. The majority of retinal telangiectasiae are secondary to local or systemic alterations such as venous occlusions and diabetes.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Telangiectasiae increased during the follow-up of the patient and became associated to retinal ischaemia areas that were not present in the baseline examination. For this reason and to avoid the development of retinal neovascularization it was decided to perform photocoagulation of said areas. Three months after photocoagulation, VA and the condition of the retina remain unchanged. Their appearance associated to CRM could be the consequence of a decompensation of the retinal hemodynamics derived from unobserved anatomical and functional alterations, which could explain the increase in telangiectasiae and ischaemia during the follow-up period. Perhaps laser treatment of said areas could have facilitated the stabilization of the condition by diminishing venous drainage requirements caused by tissue destruction.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The differential diagnostic must include retinal arteriovenous malformations (RAVM), classified by Archer (1973) and Mansour (1987). Even though Archer considered CRM as a variant of racemosa angiomas (Wyburn-Mason syndrome), they were not found in association with intracranial arteriovenous malformations. Accordingly, CRM are considered to be isolated vascular malformations of the eyes.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In the present case, the existence of arteriovenous communication could be suspected in the area temporal and close to the fovea, which could be defined as an Archer type 2. However, the characteristic appearance of the lesion led the authors to consider CRM. Generally, RAVM remain stationary although cases of spontaneous occlusion or regression has been described.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">As conclusion, even though CRM are generally asymptomatic and stable, they could give rise to visual alterations and in some cases exhibit instability that could cause the appearance of retinal complications.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflict of interests</span><p id="par0050" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres856930" "titulo" => "Abstract" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec850974" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres856929" "titulo" => "Resumen" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec850975" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case report" ] 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" => "2016-09-14" "fechaAceptado" => "2016-10-20" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec850974" "palabras" => array:3 [ 0 => "Congenital retinal macrovessel" 1 => "Retinal telangiectasia" 2 => "Retinal ischaemia" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec850975" "palabras" => array:3 [ 0 => "Macrovaso retiniano congénito" 1 => "Telangiectasias retinianas" 2 => "Isquemia retiniana" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:3 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Clinical case</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The case is presented of a 16 year-old girl with unilateral congenital retinal macrovessel associated with peripheral telangiectasia and retinal ischaemia.</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Congenital retinal macrovessel is a rare finding. To the best of our knowledge this is the first report of this unusual presentation of congenital retinal macrovessel. After 21 months of follow up, visual acuity remained stable, and no complications developed.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] "es" => array:3 [ "titulo" => "Resumen" "resumen" => "<span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Caso clínico</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de una paciente de 16 años con un macrovaso retiniano congénito unilateral, con telangiectasias retinianas periféricas asociadas a zonas de isquemia.</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discusión</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Los macrovasos retinianos congénitos constituyen un hallazgo raro, no habiéndose descrito casos asociados a telangiectasias retinianas e isquemia periférica. Tras 21 meses de seguimiento, la paciente se mantiene estable, conservando la visión inicial y sin complicaciones adicionales.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Medina-Tapia A, Molina-Sócola FE, Llerena-Manzorro L, López-Herrero F, Castilla-Martino M, Martínez-Borrego A, et al. Macrovaso retiniano asociado a telangiectasias periféricas retinianas e isquemia retiniana. Arch Soc Esp Oftalmol. 2017;92:338–342.</p>" ] ] "multimedia" => array:4 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2125 "Ancho" => 1400 "Tamanyo" => 346156 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Retinographs (a and b), RE (c) and LE (b, d–f) angiofluorescence; (d) shows a large diameter inferior temporal vein with vascular tortuosity and macular branch passing through the middle raphe and arterial tortuosity with macular branches; (e and f) presence of some telangiectasiae in the periphery.</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" => 887 "Ancho" => 1900 "Tamanyo" => 413105 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">LE angio-OCT. No alterations except in the presence of the macro vessel.</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" => 1575 "Ancho" => 1500 "Tamanyo" => 279544 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">LE angiofluorescence, showing increased telangiectasiae associated to ischaemia areas.</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" => 871 "Ancho" => 900 "Tamanyo" => 118601 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Telangiectasiae in temporal inferior area treated with laser.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Congenital retinal macrovessels" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "G.C. 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