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Surgical treatment in combined hamartoma of the retina and retinal pigment epithelium
Tratamiento quirúrgico en un hamartoma combinado de retina y epitelio pigmentario
J.L. Sánchez-Vicente, T. Rueda-Rueda, L. Llerena-Manzorro
, F.E. Molina-Socola, M. Contreras-Díaz, M. Szewc, C. Vital-Berral, A. Alfaro-Juárez, A. Medina-Tapia, F. López-Herrero, L. González-García, A. Muñoz-Morales
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Hospital Universitario Virgen del Rocío, Sevilla, Spain
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Sánchez-Vicente, T. Rueda-Rueda, L. Llerena-Manzorro, F.E. Molina-Socola, M. Contreras-Díaz, M. Szewc, C. Vital-Berral, A. Alfaro-Juárez, A. Medina-Tapia, F. López-Herrero, L. González-García, A. Muñoz-Morales" "autores" => array:12 [ 0 => array:2 [ "nombre" => "J.L." "apellidos" => "Sánchez-Vicente" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Rueda-Rueda" ] 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.E." "apellidos" => "Molina-Socola" ] 4 => array:2 [ "nombre" => "M." "apellidos" => "Contreras-Díaz" ] 5 => array:2 [ "nombre" => "M." "apellidos" => "Szewc" ] 6 => array:2 [ "nombre" => "C." "apellidos" => "Vital-Berral" ] 7 => array:2 [ "nombre" => "A." "apellidos" => "Alfaro-Juárez" ] 8 => array:2 [ "nombre" => "A." "apellidos" => "Medina-Tapia" ] 9 => array:2 [ "nombre" => "F." "apellidos" => "López-Herrero" ] 10 => array:2 [ "nombre" => "L." "apellidos" => "González-García" ] 11 => array:2 [ "nombre" => "A." "apellidos" => "Muñoz-Morales" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "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" => "Tratamiento quirúrgico en un hamartoma combinado de retina y epitelio pigmentario" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0020" "etiqueta" => "Fig. 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 729 "Ancho" => 900 "Tamanyo" => 87292 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">CHRRPE after surgery, showing diminished traction component. The macula is improved with absence of exudation, while hamartoma vessels are less tortuous. The tumor cusp is less tight.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Combined hamartoma of the retina and retinal pigment epithelium (CHRRPE) is an infrequent, benign and presumptively congenital tumor<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> that can produce significant visual acuity loss. CHRRPE expresses as a solitary and unilateral lesion located in the optic disc or posterior pole, presenting slightly raised tumors with variable amounts of pigmentation, retinal vessel tortuosity and formation of epiretinal membranes (ERM).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> In addition, CHRRPE associates a variable amount of gliosis on the retinal surface that can give rise to ERM, retinal distortion and tractional retina detachment.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The use of surgery in CHRRPE is controversial. Very few cases reporting vitrectomy for resolving the retinal traction have been published.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2–5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The case of a male, 39, who referred progressive visual acuity (VA) loss and increased metamorphopsia is presented. Vitrectomy with ERM peeling and elimination of the superficial glial component was carried out.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Clinic case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">Male, 39 years, diagnosed a few years earlier with CHRRPE in the left eye (LE), who referred progressive vision loss and increased metamorphopsia. Maximum corrected visual acuity (AVMC) was 1.0 in the right eye (RE) and 0.4 in the LE. The previous visual acuity of the LE was unknown as the patient was new, although he insisted on referring diminished vision in said eye.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The LE ocular fundus (OF) showed the presence of CHRRPE in the form of one elevated and pigmented lesion with its center in the superior temporal arch, surrounded by retina pigment epithelium (RPE) alterations, with dilated, tortuous and telangiectasic vessels, as well as a significant tractional component. Hard macular exudates were also observed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Spectral domain optic coherence tomography (SD-OCT) (Topcon<span class="elsevierStyleSup">®</span> 3D OCT-1000, Tokyo, Japan) revealed a hyper-reflective and compact mass with posterior hypo-reflective shadow, and a significant tractional component with macular involvement (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Fluorescein angiography (FAG) evidenced said vascular and RPE alterations, with slight diffusion in late times (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The rest of the exploration produced normal results.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Vitrectomy was performed, including peeling of ERM and internal limiting membrane (ILM), and extraction of a large glial component which was firmly adhered to the cusp of the tumor, achieving partial relaxation of the lesion.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Fifteen months after surgery, BCVA improved to 0.63 and metamorphopsia diminished. The appearance of the fundus and SD-OCT showed clear improvements in comparison with presurgery conditions, with diminished traction and flattening of the macula (<a class="elsevierStyleCrossRefs" href="#fig0020">Figs. 4 and 5</a>).</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">CHRRPE does not only affect the retina and the RPE as it also presents alterations in the vitreoretinal interface, producing traction and wrinkling in the retina.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The causes of diminished VA in CHRRPE include the existence of ERM, tractional changes on the fovea, retinal striations and folds, the presence of subretinal liquid and the appearance of neovascular membranes and retinal neovascularization.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">There is no consensus on the surgical treatment of CHRRPE. Whereas some authors have considered ERM as an intrinsic component of the tumor,<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">7,8</span></a> others maintain that in some cases it is an extrinsic component that is susceptible to surgery.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a> In this regard, SD-OCT has played a crucial role to decide the influence of ERM in visual loss by defining the exact location and the cleavage plane of the membrane.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">In a study carried out on 11 patients with ST-OCT, Shields et al. concluded that ERM was clearly distinct from the tumor.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a> On the basis of said studies, some authors have speculated that the ERP cells that comprise the CHRRPE migrate toward the vitreous, thus promoting the formation of ERM, which subsequently produces traction.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Accordingly, the authors believe that some patients could benefit from surgery. However, it is crucial for patients with CHRRPE to undergo close follow-up, with VA and tractional component checkups by means of SD-OCT and FAG (vascular tortuosity increases and stain diffusion).</p><p id="par0075" class="elsevierStylePara elsevierViewall">The present patient referred progressive loss of vision with increased metamorphopsia. The ERM was visible in SD-OCT, with the tractional component being evident. This led the authors to consider that vitrectomy with ERM and ILM peeling could be beneficial for the patient.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Accordingly, patients with CHRRPE having tractional components secondary to ERM and exhibiting diminished VA with increased traction would be good candidates for surgery.</p><p id="par0085" class="elsevierStylePara elsevierViewall">By way of conclusion, it should be considered that CHRRPE could account for significant vision loss. In selected patients, vitrectomy with elimination of attraction produced by ERM could achieve VA improvements.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflict of interests</span><p id="par0090" 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" => "xres807373" "titulo" => "Abstract" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec805274" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres807374" "titulo" => "Resumen" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec805273" "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" => "2016-06-08" "fechaAceptado" => "2016-07-15" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec805274" "palabras" => array:4 [ 0 => "Combined hamartoma of the retina and retinal pigment epithelium" 1 => "Epiretinal membrane" 2 => "Vitrectomy" 3 => "Optical coherence tomography" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec805273" "palabras" => array:4 [ 0 => "Hamartoma combinado de retina y epitelio pigmentario de retina" 1 => "Membrana epirretiniana" 2 => "Vitrectomía" 3 => "Tomografía de coherencia óptica" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:3 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The case is presented of a 39 year-old man with a combined hamartoma of the retina and retinal pigment epithelium, who experienced progressive visual loss and worsening of metamorphopsia. The patient underwent vitrectomy and epiretinal component peeling, with improvement in visual acuity, metamorphopsia, and retinal architecture, assessed by optical coherence tomography.</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Selected patients with combined hamartomas of the retina and retinal pigment epithelium may benefit from surgical management.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Case report" ] 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 un paciente varón de 39 años, con un hamartoma combinado de retina y epitelio pigmentario de retina. El paciente refería disminución progresiva de la visión y empeoramiento de la metamorfopsia. Se realizó una vitrectomía con pelado de la membrana epirretiniana, consiguiendo una mejoría de la agudeza visual y de la metamorfopsia, así como de la arquitectura retiniana en la tomografía de coherencia óptica.</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discusión</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Algunos pacientes seleccionados con hamartomas combinados de retina y epitelio pigmentario podrían beneficiarse de la realización de una vitrectomía.</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: Sánchez-Vicente JL, Rueda-Rueda T, Llerena-Manzorro L, Molina-Socola FE, Contreras-Díaz M, Szewc M, et al. Tratamiento quirúrgico en un hamartoma combinado de retina y epitelio pigmentario. Arch Soc Esp Oftalmol. 2017;92:137–140.</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" => 645 "Ancho" => 900 "Tamanyo" => 75695 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Left eye posterior pole retinography, showing the presence of CHRRPE in the superior zone of the macula, as an elevated mass with vascular dilatation and significant glial component on the surface. The lesion is surrounded by RPE alterations with associated hemorrhagic component and hard exudates in the macula.</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" => 1176 "Ancho" => 975 "Tamanyo" => 334984 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">SD-OCT of CHRRPE: hyper-reflective mass with posterior shadow. In 2-D image ERM can be observed whereas 3-D image shows the adherence of the vitreous to the lesion.</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" => 598 "Ancho" => 900 "Tamanyo" => 64626 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Fluorescein angiography of CHRRPE, showing RPE alteration and traction exerted over the macula.</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" => 729 "Ancho" => 900 "Tamanyo" => 87292 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">CHRRPE after surgery, showing diminished traction component. The macula is improved with absence of exudation, while hamartoma vessels are less tortuous. The tumor cusp is less tight.</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" => 903 "Ancho" => 975 "Tamanyo" => 372256 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">SD-OCT prior to surgery. Maximum thickness of 920<span class="elsevierStyleHsp" style=""></span>μm, mean thickness 424.8<span class="elsevierStyleHsp" style=""></span>μm, central thickness 853<span class="elsevierStyleHsp" style=""></span>μm, overall volume 11.98<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleSup">3</span> (top) and after surgery. Maximum thickness 409<span class="elsevierStyleHsp" style=""></span>μm, mean thickness 311.8<span class="elsevierStyleHsp" style=""></span>μm, central thickness 322<span class="elsevierStyleHsp" style=""></span>μm and overall volume 8.81<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleSup">3</span> (bottom).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Combined hamartoma of the retina and retinal pigment epithelium: findings on enhanced depth imaging optical coherence tomography in eight eyes" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "S. Arepalli" 1 => "M. Pellegrini" 2 => "S.R. Ferenczy" 3 => "C.L. 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