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BIlateral juxtapapillary retinal capillary haemangioma: Usefulness of aflibercept in the management of its complications
Hemangioma capilar retiniano yuxtapapilar bilateral: utilidad de aflibercept en el manejo de sus complicaciones
R. Campos Polo
Corresponding author
rafacampospolo@hotmail.com

Corresponding author.
, C. Rubio Sánchez, D.M. García Guisado, M.J. Díaz Luque
Servicio de Oftalmología, Hospital Virgen del Puerto, Plasencia (Cáceres), Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Clinic case report</span><p id="par0005" class="elsevierStylePara elsevierViewall">Male&#44; age 45&#44; recently diagnosed with suprarenal phaeochromocytoma&#44; who consulted due to black spot in the left eye visual field&#44; associated to metamorphopsiae with 2 days evolution&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Visual acuity &#40;VA&#41; was 1 in the right eye &#40;RE&#41; and 0&#46;7 in the left eye &#40;LE&#41;&#44; with description of paracentral metamorphopsia&#46; Anterior segment was normal and tonometry was 13<span class="elsevierStyleHsp" style=""></span>mmHg in both eyes&#46; In ocular fundus of LE&#44; a subretinal hemorrhage having 4 papillary diameters was observed adjacent to the optic nerve&#44; while the RE showed a small vascular tumor in the papillomacular bundle associated to small dot-shaped hemorrhages&#46; In fluorescein angiography&#44; the blood produced a screen effect that prevented visualization of leak area in the LE&#44; while the RE showed quick filling of the mass in arterial times that increased during the arteriovenous phase and remained stable in the late phase&#46; Optical coherence tomography &#40;OCT&#41; showed pigment epithelium detachment &#40;PED&#41; in LE at the central level of the hemorrhage as well as in the papillomacular bundle&#44; together with foveal profile rectification&#46; The RE displayed a small PED that did not alter the macular area &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; All these findings were compatible with bilateral retinal capillary hemangioma &#40;RCH&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was diagnosed with the von Hippel Lindau &#40;VHL&#41; disease and it was decided to prescribe a single aflibercept injections in the LE &#40;Eylea&#44; Reg&#233;neron&#44; Tarrytown&#44; NY&#44; USA&#44; and Bayer&#44; Leverkusen&#44; Germany&#41; as compassionate indication due to the small distance between the tumor and the optic nerve&#46; One month after the injection&#44; LE VA increased to 0&#46;8 and PED height and hemorrhage size diminished&#46; Accordingly&#44; it was decided to follow PRN treatment&#44; consisting in monthly checkups&#44; proceeding to inject if the condition improved vis-&#224;-vis the previous checkup &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; After 4 aflibercept doses &#40;in 4 consecutive months&#41;&#44; LE VA increased from 0&#46;7 to 1 &#40;without metamorphopsia&#41; and the hemorrhage resolved completely together with the PED&#46; A small contrast aggregate corresponding to the hemangioma persisted but without increasing and remaining stable in late phases &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The same occurred in the contralateral eye that did not require treatment&#46; The patient remains stable without treatment to this date&#44; with a follow-up period of 13 months&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">The presence of RCH together with some characteristic injuries &#40;such as cerebral hemangioblastoma&#44; pheochromocytoma&#41; confirmed the VHL disease<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> diagnostic&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">When said tumors are in the peripapillary area&#44; management becomes complicated above all iF VA is relatively good because treatment could produce injuries in adjacent structures&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Different guides recommend observation in asymptomatic patients where the tumor remained stable&#46; A range of treatments have been proposed including laser photocoagulation&#44; photodynamic therapy &#40;PTD&#41; or anti-VEGF treatment&#44; among others&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The efficacy of antiangiogenic treatment is due to the fact that stromal cells&#44; that form the neoplastic hemangioma component&#44; exhibit VEGF overexpression&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Fenestration in hemangioma capillaries mean that&#44; after anti-VEGF treatment&#44; contrast exhibits a localized diffusion that diminishes the exudative or hemorrhagic activity of the lesion&#46; Ach et al&#46; described good results after 9 doses of bevacizumab for treating progressive RCH located in the mid-periphery&#44; halting lesion growth&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Chelala et al&#46; obtained improvements in the size and exudation of a juxtapapillary RCH with a single ranibizumab injection&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Ziemssen et al&#46; referred positive evolution of RCH treated with bevacizumab and PTD in what concerns diminished tumor size&#44; exudation and visual acuity&#46; Said authors proposed that the association of an anti-VEGF enables a reduction of PTD treatment fluency and minimizing damages to adjacent structures&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> It has been proposed that anti-VEGF could be efficient for treating small tumors due to the higher cellular proliferation rate that is sensitive to angiogenesis inhibition&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> In addition&#44; of all proposed treatments&#44; this is the one that presents the lowest potential complications&#44; which is extremely important because the affected part is the most sensitive area of the retina&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> The authors consider that aflibercept could be a useful medicament due to its efficacy demonstrated in other retinal conditions that produce PED<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> which&#44; in the present case&#44; was due to the endophytic component of the tumor associated to significant hemorrhage&#46; In this way it was possible to control complications associated to RCH and restore patient VA by means of aflibercept monotherapy without causing damages in structures adjacent to the lesion&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Conflict of interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors&#46;</p></span></span>"
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    "fechaRecibido" => "2016-11-29"
    "fechaAceptado" => "2016-12-27"
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            0 => "Phaeochromocytoma"
            1 => "Retinal capillary haemangioma"
            2 => "Subretinal juxtapapillary hemorrhage"
            3 => "Von Hippel Lindau disease"
            4 => "Aflibercept"
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            0 => "Feocromocitoma"
            1 => "Hemangioma capilar retiniano"
            2 => "Hemorragia subretiniana yuxtapapilar"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 45 year-old man with a history of adrenal phaeochromocytoma presented with a subretinal juxtapapillary hemorrhage on his left eye and a small asymptomatic vascular tumor in the contralateral eye&#46; With the mentioned findings&#44; the patient was diagnosed with bilateral retinal capillary haemangioma in the context of a von Hippel Lindau disease&#46; Intravitreal aflibercept was prescribed&#44; with a good outcome of the disease&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Many treatments have been proposed for the management of juxtapapillary retinal capillary haemangioma with variable results&#46; Intravitreal aflibercept can be a useful treatment with a good safety profile&#46;</p></span>"
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        "resumen" => "<span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Caso cl&#237;nico</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Var&#243;n de 45 a&#241;os&#44; con antecedentes personales de feocromocitoma suprarrenal&#44; que presenta una hemorragia subretiniana yuxtapapilar en el ojo izquierdo y un peque&#241;o tumor vascular asintom&#225;tico en el ojo contralateral&#46; Con los hallazgos mencionados&#44; el paciente fue diagnosticado de hemangioma capilar retiniano bilateral en el contexto de una enfermedad de von Hippel Lindau y se paut&#243; aflibercept intrav&#237;treo&#44; con una buena evoluci&#243;n del cuadro&#46;</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discusi&#243;n</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Se han propuesto m&#250;ltiples tratamientos para el manejo del hemangioma capilar retiniano yuxtapapilar&#44; con unos resultados variables&#46; El aflibercept intrav&#237;treo puede ser un tratamiento de utilidad&#44; con un buen perfil de seguridad&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Campos Polo R&#44; Rubio S&#225;nchez C&#44; Garc&#237;a Guisado DM&#44; D&#237;az Luque MJ&#46; Hemangioma capilar retiniano yuxtapapilar bilateral&#58; utilidad de aflibercept en el manejo de sus complicaciones&#46; Arch Soc Esp Oftalmol&#46; 2017&#59;92&#58;477&#8211;480&#46;</p>"
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          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">RE images&#58; &#40;a&#41; retinograph&#44; &#40;c&#41; angiograph&#44; &#40;e&#41; OCT&#46; LE images&#58; &#40;b&#41; retinograph&#44; &#40;d&#41; angiograph &#40;screen effect of subretinal hemorrhage preventing visualization of RCH&#41;&#44; &#40;f&#41; OCT &#40;subretinal hemorrhage produces PED&#41;&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">LE images after the first aflibercept injection&#58; &#40;a&#41; retinograph&#44; &#40;d&#41; angiograph&#44; &#40;g&#41; OCT&#46; After the 2nd injection&#58; &#40;b&#41; retinograph&#44; &#40;e&#41; angiograph&#44; &#40;h&#41; OCT&#46; After the 3rd injection&#58; &#40;c&#41; retinograph&#44; &#40;f&#41; angiograph&#44; &#40;i&#41; OCT&#46; Improvement is notable&#46;</p>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">&#40;a&#41; Retinograph&#44; &#40;b&#41; angiograph&#44; &#40;c&#41; macular surface map&#44; and &#40;d&#41; LE OCT one month after the 4th aflibercept injection&#46; Hemorrhage and PED have disappeared&#44; with small contrast aggregate persisting in angiograph&#46;</p>"
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Article information
ISSN: 21735794
Original language: English
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