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Epithelial inclusion cyst secondary to glaucoma surgery: A case series
Quiste de inclusión epitelial secundario a cirugía de glaucoma: serie de casos
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"apellidos" => "Quilez Larragan" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0365669124000893" "doi" => "10.1016/j.oftal.2024.05.003" "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/S0365669124000893?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579424001129?idApp=UINPBA00004N" "url" => "/21735794/0000009900000008/v1_202408010614/S2173579424001129/v1_202408010614/en/main.assets" ] "en" => array:19 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Epithelial inclusion cyst secondary to glaucoma surgery: A case series" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "356" "paginaFinal" => "358" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "B. Benito-Pascual, O. Rodríguez-Quet, C. Méndez-Hernández, L. Álvarez-Cascos-López, L. Gil-Amado, J. García-Feijoo" "autores" => array:6 [ 0 => array:4 [ "nombre" => "B." "apellidos" => "Benito-Pascual" "email" => array:1 [ 0 => "blancabenito91@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "O." "apellidos" => "Rodríguez-Quet" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Méndez-Hernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Álvarez-Cascos-López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "L." "apellidos" => "Gil-Amado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 5 => array:3 [ "nombre" => "J." "apellidos" => "García-Feijoo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Unidad de Córnea, Servicio de Oftalmología, Hospital Universitario Severo Ochoa, Leganés, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Unidad de Glaucoma, Servicio de Oftalmología, Hospital Clínico San Carlos, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Quiste de inclusión epitelial secundario a cirugía de glaucoma: serie de casos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 768 "Ancho" => 768 "Tamanyo" => 87364 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Development of epithelial inclusion cyst in the months prior to removal of the epithelial inclusion.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">In certain situations the stratified squamous epithelium of the ocular surface (originating from the cornea or conjunctiva) may grow into the eye extending through the anterior segment structures.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The epithelial cells can grow in the form of "sheets" or form cysts. The latter are called epithelial inclusion cysts (EIC).</p><p id="par0010" class="elsevierStylePara elsevierViewall">EICs were first described by Rothmund in 1872. They were defined as surface epithelial growths on the iris secondary to penetrating wounds of the eyeball.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> There are two clinical forms of EICs described in the literature: the pearly form when they are opaque with a pearly sheen, and the serous form when they are transparent and contain slightly cloudy fluid.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Since they were first published, cases have been described in cataract surgery, penetrating keratoplasty and trauma.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Although infrequent, the consequences of intraocular growth of EICs can be as devastating as glaucoma, due to the space compromise they generate. Over the years, several surgical approaches have been developed for their treatment such as simple aspiration, with high recurrence rates, or aspiration with sclerosing agent which, although more effective, presents the associated risk of toxicity due to extravasation of the sclerosing substance. Therefore, the recommended treatment today is total surgical excision of the lesions.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Glaucoma surgeries include trabeculectomy, drainage implants, MIGS (Minimally Invasive Glaucoma Surgery) and MIBS (Minimally Invasive Bleb Surgery), among others. To date, there is no evidence described in the literature of CIS secondary to these surgeries. We present 2 cases of CIE secondary to trabeculectomy and PRESERFLO® MicroShunt (Santen, Osaka, Japan).</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case series</span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Case 1</span><p id="par0030" class="elsevierStylePara elsevierViewall">A 79-year-old patient with moderate-severe progressive chronic open-angle glaucoma, more severe in the left eye, who underwent cataract extraction plus trabeculectomy in the left eye in 2013. Two years later, after failure of trabeculectomy, an Ahmed valve implant was necessary in the same eye, with good intraocular pressure (IOP) control to date, with no need for medical treatment. In 2014, cataract extraction was performed in the right eye. In 2016, IOP started to go out of control in this eye, and after rejecting the trabeculectomy option due to failure in the left eye, it was decided to use the PRESERFLO® MicroShunt implant (Santen, Osaka, Japan). In the postoperative period, contact with the endothelium was detected. During surgery to reposition the device, a major hemorrhage due to the patient's systemic anticoagulant treatment for his risk of cardiovascular event prevented repositioning of the device and it was decided to remove it. Due to the good control of intraocular pressure, no further surgeries were required and ocular pressure was controlled without topical treatment. The cause of the normalization of ocular pressure is unknown; it is hypothesized that the drainage pathway remained permeable even in the absence of the device. In 2019, an EIC was observed in the right eye with a serous appearance (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient underwent ultrasonic biomicroscopy (UBM) which confirmed the benign nature of the lesion. The patient is being followed up by the glaucoma department and to date no cyst growth has been detected, with IOP controlled without further interventions or topical medication. Therefore no new interventions have been required.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Case 2</span><p id="par0035" class="elsevierStylePara elsevierViewall">A 9-year-old female with glaucoma in the left eye secondary to uveitis due to juvenile idiopathic arthritis (JIA). Due to poor IOP control with topical medications, an Ahmed valve implant was performed in 2014. Two years after implantation, she developed an EIC in the uvea below the tube which had to be surgically removed (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Subsequently, it was necessary to reposition the tube on 2 occasions. To date, the EIC has not recurred, with IOP and uveitis remaining under control.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Although epithelial growth after glaucoma drainage device implantation cases have been published in the literature, none describe EIC.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The development of EIC rather than the lamellar form, which consists of sheets of epithelium within the eye, is still unclear. According to published case reports, cyst development takes weeks to years from the surgery or injury that causes the epithelial cells to penetrate into the eye. The most widely accepted theory is that the epithelium remains intact until an inflammatory stimulus activates the cells and the cyst proliferates.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> After considering other less invasive treatments, the <span class="elsevierStyleItalic">gold standard</span> for the treatment of this type of lesion is surgery, by complete excision of the lesion, as this technique has a lower regression rate than simple aspiration and a lower rate of uncontrolled complications than sclerosing agents.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Due to the anatomical repercussions and the intrinsic risk of surgery, the intervention is reserved for dangerous cases, while follow-up of all patients is mandatory. A key aspect in the management of these patients is the rapid identification of the lesion in order to anticipate possible fatal repercussions and to be able to perform an early surgical approach, with less mutilating interventions.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">To provide adequate patient care, a differential diagnosis with congenital epithelial cysts (intraocular choristoma) is needed. The clinical history (absence of wounds) is the main key to the differential diagnosis, since the histology is similar.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In the first case described, epithelial cells could have penetrated into the anterior chamber during implantation of PRESERFLO® MicroShunt (Santen, Osaka, Japan), and the inflammatory blood stimulus after removal of the device could have activated the cells to form the cyst. In this case, only follow-up is needed, as rushing to unnecessary early surgical removal could lead to excessive risk, not offset by the lack of benefit from surgery at this time.</p><p id="par0055" class="elsevierStylePara elsevierViewall">In the second case, epithelial cells may have penetrated the anterior chamber during Ahmed valve implantation and the inflammatory stimulus of JIA may have activated the cells. In this case, the cyst interfered with the aqueous humor drainage system, requiring surgical removal. Finally, histology confirmed the epithelial origin of the lesion.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conclusion</span><p id="par0060" class="elsevierStylePara elsevierViewall">Ophthalmologists should be alert to this type of pathology. They should recognize it by its clinical appearance and determine the cause that produced the penetration of epithelial cells and the activation stimulus. It is important to know that this is a benign proliferation, but it requires surgical removal when complications occur, and it is important to follow up patients to anticipate this type of complications in order to achieve early removal, with less complicated surgery and better results. Finally, histologic confirmation is essential for a proper diagnosis.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Ethical considerations</span><p id="par0065" class="elsevierStylePara elsevierViewall">Research Ethics Committee of the Hospital Clínico San Carlos.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Funding</span><p id="par0070" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflict of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:12 [ 0 => array:3 [ "identificador" => "xres2214471" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1856576" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres2214472" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1856575" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:3 [ "identificador" => "sec0010" "titulo" => "Case series" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0015" "titulo" => "Case 1" ] 1 => array:2 [ "identificador" => "sec0020" "titulo" => "Case 2" ] ] ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0035" "titulo" => "Ethical considerations" ] 9 => array:2 [ "identificador" => "sec0040" "titulo" => "Funding" ] 10 => array:2 [ "identificador" => "sec0045" "titulo" => "Conflict of interest" ] 11 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2024-02-18" "fechaAceptado" => "2024-05-05" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1856576" "palabras" => array:3 [ 0 => "Glaucoma" 1 => "Drainage implant" 2 => "Epithelial cysts" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1856575" "palabras" => array:3 [ 0 => "Glaucoma" 1 => "Implante de drenaje" 2 => "Quiste epitelial" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Epithelial inclusion cysts (EIC) are a rare ocular disease and its physiopathology is not well-known. They consist on growths of ocular surface epithelial cells inside the anterior segment of the eye in the form of a cyst. To date several cases have been published in the literature, none of them related to glaucoma surgery. We describe two cases of EIC after glaucoma devices implantation. An 86 year-old male patient with primary open angle glaucoma develop an EIC in right eye three years after removal of PRESERFLO™ MicroShunt (Santen, Osaka, Japan) and a 9 year-old female patient with glaucoma secondary to uveitis for juvenile idiopathic arthritis develops an EIC under the tube of an Ahmed valve implant during postoperative period. EIC develop after ocular penetrating wounds and an inflammatory stimulus. They are benign proliferations, follow-up is necessary to detect space complications early, so less mutilating surgery is needed for removal.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Los quistes de inclusión epitelial (QIE) representan una patología ocular muy poco frecuente con una fisiopatología aún no del todo conocida. Consisten en el crecimiento de células epiteliales de la superficie ocular dentro del segmento anterior del ojo en forma de quistes. Aunque se han publicado casos en la literatura sobre este tipo de lesiones, ninguno de ellos se ha relacionado con la cirugía de glaucoma hasta la fecha. Describimos dos casos de QIE después del implante de dispositivos de drenaje de glaucoma. El primer caso se trata de un varón de 86 años con glaucoma primario de ángulo abierto que desarrolló un QIE tres años después de la extracción de PRESERFLO® MicroShunt, y el segundo, una mujer de 9 años con glaucoma secundario a uveítis por artritis idiopática juvenil que desarrolló un QIE debajo del tubo de la válvula Ahmed en el postoperatorio. Los QIE son proliferaciones benignas que se desarrollan tras heridas penetrantes o estímulos inflamatorios importantes. Requieren seguimiento para detectar complicaciones tempranas y evitar posteriores cirugías mutilantes en caso de ser necesaria su extracción.</p></span>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1000 "Ancho" => 1300 "Tamanyo" => 177183 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Case 1. A and B: PRESERFLO® MicroShunt (Santen, Osaka, Japan). C and D: epithelial inclusion cyst after removal of PRESERFLO® MicroShunt (Santen, Osaka, Japan).</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 768 "Ancho" => 768 "Tamanyo" => 87364 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Development of epithelial inclusion cyst in the months prior to removal of the epithelial inclusion.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Epithelial downgrowth. Report of 2 cases diagnosed by ocular biopsy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "H.R. Patel" 1 => "C.E. Margo" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.anndiagpath.2016.10.004" "Revista" => array:6 [ "tituloSerie" => "Ann Diagn Pathol" "fecha" => "2017" "volumen" => "26" "paginaInicial" => "60" "paginaFinal" => "63" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28209236" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Ueber cysten der Regenbogenhaut" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "A. 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Margo" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1001/archopht.1997.01100150670021" "Revista" => array:7 [ "tituloSerie" => "Arch Ophthalmol" "fecha" => "1997" "volumen" => "115" "paginaInicial" => "668" "paginaFinal" => "669" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/9152138" "web" => "Medline" ] ] "itemHostRev" => array:3 [ "pii" => "S0301211519302337" "estado" => "S300" "issn" => "03012115" ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Epithelial downgrowth after Ahmed implantation presenting as a peritubular fibrovascular membrane" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:7 [ 0 => "J. Hu" 1 => "D.A. Goldstein" 2 => "Y.I. Leiderman" 3 => "V. Malhotra" 4 => "F.Y. Chau" 5 => "A.Y. Lin" 6 => "T.S. Vajaranant" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1097/ijg.0b013e318255d9ee" "Revista" => array:6 [ "tituloSerie" => "J Glaucoma" "fecha" => "2013" "volumen" => "22" "paginaInicial" => "e11" "paginaFinal" => "e13" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23899696" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735794/0000009900000008/v1_202408010614/S2173579424001130/v1_202408010614/en/main.assets" "Apartado" => array:4 [ "identificador" => "5812" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Short communications" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735794/0000009900000008/v1_202408010614/S2173579424001130/v1_202408010614/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579424001130?idApp=UINPBA00004N" ]
