array:24 [ "pii" => "S2173579419300349" "issn" => "21735794" "doi" => "10.1016/j.oftale.2018.12.008" "estado" => "S300" "fechaPublicacion" => "2019-05-01" "aid" => "1454" "copyright" => "Sociedad Española de Oftalmología" "copyrightAnyo" => "2019" "documento" => "article" "crossmark" => 1 "subdocumento" => "fla" "cita" => "Arch Soc Esp Oftalmol. 2019;94:248-51" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "Traduccion" => array:1 [ "es" => array:19 [ "pii" => "S0365669119300206" "issn" => "03656691" "doi" => "10.1016/j.oftal.2018.12.004" "estado" => "S300" "fechaPublicacion" => "2019-05-01" "aid" => "1454" "copyright" => "Sociedad Española de Oftalmología" "documento" => "article" "crossmark" => 1 "subdocumento" => "fla" "cita" => "Arch Soc Esp Oftalmol. 2019;94:248-51" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:2 [ "total" => 30 "formatos" => array:2 [ "HTML" => 20 "PDF" => 10 ] ] "es" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Comunicación corta</span>" "titulo" => "Xantogranuloma orbitario del adulto como causa de inflamación palpebral recidivante" "tienePdf" => "es" "tieneTextoCompleto" => "es" "tieneResumen" => array:2 [ 0 => "es" 1 => "en" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "248" "paginaFinal" => "251" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Adult orbital xanthogranuloma as a cause of recurrent palpebral swelling" ] ] "contieneResumen" => array:2 [ "es" => true "en" => true ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 760 "Ancho" => 950 "Tamanyo" => 75153 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">A: aspecto clínico de la paciente en la primera visita. Muestra un engrosamiento del PSD (flecha). B: aspecto clínico de la paciente tras una reagudización del cuadro, con aumento del tamaño de la masa y rubor en PSD (flecha).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "E. Navarro-Hernández, A. Hernández-Pons, S. Montolío-Marzo, M. Pérez-López" "autores" => array:4 [ 0 => array:2 [ "nombre" => "E." "apellidos" => "Navarro-Hernández" ] 1 => array:2 [ "nombre" => "A." "apellidos" => "Hernández-Pons" ] 2 => array:2 [ "nombre" => "S." "apellidos" => "Montolío-Marzo" ] 3 => array:2 [ "nombre" => "M." "apellidos" => "Pérez-López" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S2173579419300349" "doi" => "10.1016/j.oftale.2018.12.008" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579419300349?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0365669119300206?idApp=UINPBA00004N" "url" => "/03656691/0000009400000005/v2_202201280719/S0365669119300206/v2_202201280719/es/main.assets" ] ] "itemSiguiente" => array:19 [ "pii" => "S2173579419300088" "issn" => "21735794" "doi" => "10.1016/j.oftale.2018.10.013" "estado" => "S300" "fechaPublicacion" => "2019-05-01" "aid" => "1427" "copyright" => "Sociedad Española de Oftalmología" "documento" => "article" "crossmark" => 1 "subdocumento" => "fla" "cita" => "Arch Soc Esp Oftalmol. 2019;94:252-6" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Study of aggressive carcinomas through orbital metastasis" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "252" "paginaFinal" => "256" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Estudio de carcinomas agresivos a través de metástasis orbitarias" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "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" => 1653 "Ancho" => 2083 "Tamanyo" => 285700 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Case 2. (A and B) External examination with loss of conjugated gaze, ptosis and incongruent ocular movements. Orbit CT (C) showing bilateral metastatic infiltration of both horizontal rectus muscles.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "G. Espinosa-Barberi, C. Alba Linero, F.J. Galván González, E. Álvarez González, A. Rey López, F. Medina Rivero" "autores" => array:6 [ 0 => array:2 [ "nombre" => "G." "apellidos" => "Espinosa-Barberi" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Alba Linero" ] 2 => array:2 [ "nombre" => "F.J." "apellidos" => "Galván González" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Álvarez González" ] 4 => array:2 [ "nombre" => "A." "apellidos" => "Rey López" ] 5 => array:2 [ "nombre" => "F." "apellidos" => "Medina Rivero" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0365669118303368" "doi" => "10.1016/j.oftal.2018.10.012" "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/S0365669118303368?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579419300088?idApp=UINPBA00004N" "url" => "/21735794/0000009400000005/v1_201904290704/S2173579419300088/v1_201904290704/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S2173579419300404" "issn" => "21735794" "doi" => "10.1016/j.oftale.2019.01.006" "estado" => "S300" "fechaPublicacion" => "2019-05-01" "aid" => "1468" "copyright" => "Sociedad Española de Oftalmología" "documento" => "article" "crossmark" => 1 "subdocumento" => "fla" "cita" => "Arch Soc Esp Oftalmol. 2019;94:242-7" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Periorbital necrotising fasciitis secondary to scratching" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "242" "paginaFinal" => "247" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Fascitis necrosante periorbitaria secundaria a rascado" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1114 "Ancho" => 1305 "Tamanyo" => 232224 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Photocomposition: (A) axial CT section at the upper eyelid level showing edematization of predominantly preseptal periorbital soft tissues. (B) Axial CT section at the lower eyelid level showing periorbital tissues edema at the preseptal level. (C) CT coronal section showing edematization of periorbital soft tissues in the external canthus and right cheek. (D) Right eye eyelid photography showing fibrinoid necrosis plaques in both eyelids. (E) Appearance after surgical debridement of areas of fibrinoid necrosis and betadine net lining. (The authors have the informed consent of the patient for publishing the images that illustrate this article.).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "I.A. Placinta, E. España-Gregori, A. Rodrigo-Hernández, C. Martínez-Rubio, J. Safont-Albert, M.Á. Bort-Martí" "autores" => array:6 [ 0 => array:2 [ "nombre" => "I.A." "apellidos" => "Placinta" ] 1 => array:2 [ "nombre" => "E." "apellidos" => "España-Gregori" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Rodrigo-Hernández" ] 3 => array:2 [ "nombre" => "C." "apellidos" => "Martínez-Rubio" ] 4 => array:2 [ "nombre" => "J." "apellidos" => "Safont-Albert" ] 5 => array:2 [ "nombre" => "M.Á." "apellidos" => "Bort-Martí" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0365669119300346" "doi" => "10.1016/j.oftal.2019.01.005" "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/S0365669119300346?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579419300404?idApp=UINPBA00004N" "url" => "/21735794/0000009400000005/v1_201904290704/S2173579419300404/v1_201904290704/en/main.assets" ] "en" => array:20 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Adult orbital xanthogranuloma as a cause of recurrent palpebral swelling" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "248" "paginaFinal" => "251" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E. Navarro-Hernández, A. Hernández-Pons, S. Montolío-Marzo, M. Pérez-López" "autores" => array:4 [ 0 => array:3 [ "nombre" => "E." "apellidos" => "Navarro-Hernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Hernández-Pons" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:4 [ "nombre" => "S." "apellidos" => "Montolío-Marzo" "email" => array:1 [ 0 => "s.montoliomarzo@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 3 => array:3 [ "nombre" => "M." "apellidos" => "Pérez-López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Virgen de los Lirios, Alcoy, Alicante, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario Dr. Peset, Valencia, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Oftalmología, Unidad de Órbita, Párpados y Vía Lagrimal, FISABIO Oftalmología Médica, Valencia, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Xantogranuloma orbitario del adulto como causa de inflamación palpebral recidivante" ] ] "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" => 1070 "Ancho" => 950 "Tamanyo" => 73026 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Optical coherence tomography showing preseptal infiltration in the right eye (arrow). (A) Axial projection. (B) Lateral projection.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Adult orbital and adnexal xanthogranulomatous diseases comprise a heterogeneous group of infrequent idiopathic inflammatory disorders that can be classified in 4 groups<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a>: adult xanthogranuloma, adult periocular xanthogranuloma associated with asthma, necrobiotic xanthogranuloma and Erdheim-Chester disease. At the clinic level, orbital or periocular inflammatory lesions are associated to subcutaneous yellowish nodules which in some cases could be difficult to differentiate from highly frequent palpebral lesions such as palpebral xanthelasm (XP). Adequate identification of xanthogranulomatous lesions and differential diagnostic with PX is very important for possible associations with systemic paraneoplastic disease of the former. All xanthogranulomatous diseases are histologically characterized by infiltration of xanthomized histiocytes and Touton-type giant multi-nucleated cells in affected tissue, although it is possible to find ultrastructural differences between them.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">An adult xanthulogranuloma in anterior orbital location is presented. The case debuted with repeated palpebral edema episodes without the typical yellowish infiltration characterizing the entity, thus increasing the difficulty of the baseline diagnostic.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinic case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">Female, 43, who visited due to repeated inflammation in the right upper eyelid (RUE) with subjective tissue thickening. The patient referred dermatochalasis in upper eyelids since 2009 after gestation. She referred undergoing esthetic blepharoplasty surgery in 2011 and again in 2015 due to residual dermatochalasis. In addition, she referred palpebral thickening with recurring edema episodes since the first palpebral surgery.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Physical examination produced a corrected best visual acuity of 1 in both eyes (BE) and normal chromatic vision. Exophthalmometry was 18<span class="elsevierStyleHsp" style=""></span>mm in BE without spontaneous diplopia in any gaze position. Palpation enabled the identification of a painless hard mass in the fixed portion of the RUE that extended along and beyond the limits of the previous blepharoplasty scar that was not adhered to deep planes (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Periocular examination was normal in the left eye. Palpebral color was normal in BE and symmetrical in the upper and lower eyelids. The palpebral opening in BE was normal (9<span class="elsevierStyleHsp" style=""></span>mm), the upper margin-reflex distance was 4<span class="elsevierStyleHsp" style=""></span>mm and the palpebral groove was located at 7<span class="elsevierStyleHsp" style=""></span>mm. Biomicroscopy and funduscopy examination in BE was normal. Considering the findings, magnetic resonance was requested which evidenced diffuse soft tissue thickening compatible with cicatricial tissue in the RUE.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">During the follow-up, the patient developed an episode of painless palpebral edema and hyperemia in the RUE, with increased thickening of the subcutaneous mass and hardening upon palpation, accompanied by inferior palpebral edema (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Examination did not evidence signs of post-septal orbital compromise, while pupil examination, chromatic vision and extrinsic ocular motility produced normal results. An urgent computerized tomography was taken that showed pre-septal infiltration (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) giving rise to differential diagnostic between granuloma due to foreign body, hypertrophic scar, lymphoid hyperplasia or possible lymphoma. Anterior orbital biopsy was scheduled (through upper palpebral groove) which identified pre-septal rubber-like hard whitish tissue. Intra-surgery analysis of part of the sample discarded the presence of neoplastic cells. The identified pathological tissue was removed, including indurated tissue which was referred for definitive analysis. Postoperatively, oral prednisone was prescribed at an initial dose of 60<span class="elsevierStyleHsp" style=""></span>mg/day in descending pattern during 2 weeks.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The anatomopathological study showed cellular proliferation with diffuse distribution of xanthomized histiocytes associated to mature lymphocytes and giant multinucleated cells with Touton-type coronal nuclei (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). No signs of necrosis, vasculitis or malignity were found. Said findings were diagnosed as histiocytic xanthogranuloma. It was not necessary to carry out immunohistochemistry techniques for characterizing the lesion.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">The patient was requested to provide photographs taken before the esthetic blepharoplasties, which showed yellowish colors in both upper eyelids, possibly diagnosed as PX without associated histological study.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The Medicine Dept. performed a systemic examination and an extension study that included proteingram and blood determination of beta-2-microglobulin, with all parameters producing normal results. This excluded possible associated monoclonal gammapathy. No lymph adenopathies or respiratory clinic were evidenced. In the absence of systemic compromise and the presence of a solitary lesion, the condition was diagnosed as adult orbital xanthogranuloma.</p><p id="par0045" class="elsevierStylePara elsevierViewall">At present, the patient remained asymptomatic without treatment and with biannual checkups, without clinic signs of asthma or pathologic hematological findings. In addition, she has not exhibited new periocular inflammation episodes.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">The adult orbital xanthogranulomatous disease comprises a heterogeneous group of infrequent inflammatory idiopathic diseases characterized by proliferation of localized or systemic non-Langerhans histiocytes. Participating etiopathogenic mechanisms are not yet exactly known.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> Said disease generally affects middle-aged adults and can debut with yellowish subcutaneous, subconjunctival or periocular infiltrations with associated mass effect having variable magnitudes.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> Said yellowish skin infiltration is present in 72% of patients, including the 4 variants of the spectrum. It can also cause other ocular symptoms such as iritis, keratitis, diplopia, exophthalmos and blindness.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">When suspecting a periocular xanthogranulomatous lesion it is important to take a biopsy and make a histopathological study as well as a systemic extension study in order to discard clinic entities that could be associated and jeopardize the life of the patient. On the other hand, therapeutic management remains highly controversial due to the scarcity of available scientific evidence (grade C).<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In said group of xanthogranulomatous disorders, adult xanthogranuloma is infrequently discussed in the literature. Sivak-Callcott et al. found a total amount of 8 documented cases.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> It appears in the age range of 17–85 years, without predilection for sex and debuts with an isolated lesion without systemic compromise. It is generally limited to the orbit, mainly anterior/preseptal and has a positive prognostic. As regards treatment, surgical exeresis is regarded as the best option,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> possibly combined with adjuvant therapies such as corticoids.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> Observation can also be considered.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> However, even though the patient does not exhibit additional symptoms, it is necessary to discard other diseases that are associated to the rest of orbital inflammatory disorders.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Periocular xanthogranuloma associated to asthma in the adult could associate late onset asthma as well as lymphadenopathies. In these cases, the prognostic is also positive and a good treatment option could be surgical exeresis associated to corticoid therapy.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">The initial clinic of necrobiotic xanthogranuloma is skin ulceration followed by fibrosis. It is frequently associated to IgG monoclonal gammapathy in 80% of cases and in lower proportion to multiple myeloma and cryoglobulinemia type 1. In these patients, clinic supervision with repetition proteingrams is essential. A good option for treatment is the combination of corticoids with immunosuppressant agents but even so the prognostic is generally negative.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Finally, the Erdheim-Chester disease debuts with compromise of the orbit and multiple organs such as the heart, lungs, retroperitoneum and bones. Bone lesions are visible in radiological studies (lytic destruction of long bones) and are pathognomic of said entity. Due to the compromise of vital organs the prognosis is negative with a mortality in the area of 60%.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">In the present patient, the previous blepharoplasty surgeries possibly extirpated the upper eyelid xanthochromic tissue evidenced in the presurgery photographs. The anatomopathological analysis of lesions initially classified as PX can identify orbital xanthogranulomas in some cases.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> The existence of nodular components, diffuse compromise (not limited to eyelids, extension to adjacent skin or ulceration) and induration associated to tissue must lead to the suspicion of diseases other than a simple PX.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> An additional peculiarity of the present case is the recurrent palpebral edema associated to induration which is not described in the literature as a form of presentation of adult xanthogranuloma.</p><p id="par0085" class="elsevierStylePara elsevierViewall">The only treatment necessary for the present patient was surgical exeresis of the lesion because after xanthogranuloma resection she did not exhibit new inflammation episodes. Corticoid or immunosuppressant treatment is indicated for patients when it is not possible to extirpate the lesion, the extirpation is incomplete or inflammatory clinic persists.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0090" class="elsevierStylePara elsevierViewall">Periocular/orbital xanthogranulomas, sometimes regarded as “malign xanthelasmas”, are a rare entity which is potentially severe and difficult to diagnose due to its low incidence and variable clinic presentation. The ophthalmologist should be acquainted with this entity. It is recommendable to perform a histological analysis of the lesions which clinically could appear as the palpebral xanthelasmas. After the definitive diagnostic, an extension study must be performed with the aim of discarding potentially fatal systemic compromise.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflict of interests</span><p id="par0095" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1184812" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1104922" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1184813" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1104923" "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" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-10-23" "fechaAceptado" => "2018-12-18" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1104922" "palabras" => array:3 [ 0 => "Xanthogranuloma" 1 => "Adult orbital xanthogranulomatous disease" 2 => "Orbital inflammation" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1104923" "palabras" => array:3 [ 0 => "Xantogranuloma" 1 => "Xantogranuloma orbitario del adulto" 2 => "Inflamación orbitaria" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Orbital adult-onset xanthogranuloma is a rare idiopathic inflammatory disorder included in the group of orbital xanthogranulomatous diseases with possible systemic associations.</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A 43 year-old woman presented with a deep fast-growing mass on the right upper eyelid. She had a past medical history of bilateral non-ulcerative nodules on the upper eyelid and two cosmetic upper lid blepharoplasties. An excisional biopsy was performed and the histopathology analysis confirmed the diagnosis of adult histiocytic xanthogranuloma. The systemic work up was negative and the patient is asymptomatic without treatment.</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Orbital xanthogranulomatous diseases are rare, but potentially life-threatening disorders. The ophthalmologist must be aware of this disease in order to make an early diagnosis using the anamnesis, clinical examination, and histology of the periocular lesions that may resemble palpebral xanthelasmas.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El xantogranuloma orbitario del adulto es un raro trastorno inflamatorio idiopático incluido dentro de las enfermedades xantogranulomatosas orbitarias, con posible asociación a otras condiciones sistémicas.</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de una mujer de 43 años que presentó una masa profunda de crecimiento rápido en el párpado superior derecho con antecedentes de nódulos amarillentos no ulcerativos en el párpado superior, bilaterales, y 2 blefaroplastias superiores previas. Se realizó biopsia-extirpación de la lesión orbitaria anterior, siendo los hallazgos anatomopatológicos compatibles con xantogranuloma histiocítico del adulto. El estudio sistémico fue negativo y la paciente se encuentra asintomática sin tratamiento.</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Las enfermedades xantogranulomatosas orbitarias constituyen una entidad rara potencialmente grave, por lo que el oftalmólogo debe de estar familiarizado con esta enfermedad para poder realizar un diagnóstico precoz a través de la anamnesis, exploración y la histología de lesiones que en ocasiones pueden simular xantelasmas palpebrales.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Navarro-Hernández E, Hernández-Pons A, Montolío-Marzo S, Pérez-López M. Xantogranuloma orbitario del adulto como causa de inflamación palpebral recidivante. Arch Soc Esp Oftalmol. 2019;94:248–251.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 760 "Ancho" => 950 "Tamanyo" => 75153 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">(A) Patient clinic at the first visit, showing thickening of RUE (arrow). (B) Patient clinic after condition worsening, with increased mass size and reddening in RUE (arrow).</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" => 1070 "Ancho" => 950 "Tamanyo" => 73026 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Optical coherence tomography showing preseptal infiltration in the right eye (arrow). (A) Axial projection. (B) Lateral projection.</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" => 976 "Ancho" => 1500 "Tamanyo" => 341675 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">Anatomopathological study of the analyzed sample. (A) Macroscopic sample photograph showing whitish fragments with uneven shape and surface measuring 1.3<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>0.7<span class="elsevierStyleHsp" style=""></span>cm, 1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>0.3<span class="elsevierStyleHsp" style=""></span>cm, 0.6<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>0.2<span class="elsevierStyleHsp" style=""></span>cm and 0.5<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>0.5<span class="elsevierStyleHsp" style=""></span>cm, respectively. (B) Histological section with hematoxylin–eosine staining (400<span class="elsevierStyleHsp" style=""></span>×) showing diffuse a cellular proliferation comprising xanthomized histiocytes (*) associated to a small mature lymphocyte component making small aggregates (+). (C) Focal image (200×) showing giant multi-nucleated cells with Touton-type coronal nuclei (*). No areas of necrosis, vasculitis or malignity can be observed.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult orbital and adnexal xanthogranulomatous disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "M.J. Davies" 1 => "K. Whitehead" 2 => "G. Quagliotto" 3 => "D. Wood" 4 => "R.S. Patheja" 5 => "T.J. Sullivan" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Asia Pac J Ophthalmol" "fecha" => "2017" "volumen" => "6" "paginaInicial" => "435" "paginaFinal" => "443" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0045" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult orbital xanthogranulomatous disease: a review with emphasis on etiology, systemic associations, diagnostic tools, and treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J. Kerstetter" 1 => "J. Wang" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Dermatol Clin" "fecha" => "2015" "volumen" => "33" "paginaInicial" => "457" "paginaFinal" => "463" ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0050" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult xanthogranulomatous disease of the orbit and ocular adnexa: new immunohistochemical findings and clinical review" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "J.A. Sivak-Callcott" 1 => "J. Rootman" 2 => "S.L. Rasmussen" 3 => "R.A. Nugent" 4 => "V.A. White" 5 => "D. Paridaens" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1136/bjo.2005.085894" "Revista" => array:6 [ "tituloSerie" => "Br J Ophthalmol" "fecha" => "2006" "volumen" => "90" "paginaInicial" => "602" "paginaFinal" => "608" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/16622091" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0055" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult orbital xanthogranulomatous disease: adult-onset xanthogranuloma of periorbital location" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "M. Minami-Hori" 1 => "I. Takahashi" 2 => "M. Honma" 3 => "Y. Ito" 4 => "H. Takahashi" 5 => "A. Ishida-Yamamoto" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Clin Exper Dermatol" "fecha" => "2011" "volumen" => "36" "paginaInicial" => "628" "paginaFinal" => "631" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0060" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult orbital xanthogranulomas: clinical features and management" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "H. Kiratli" 1 => "M. Kilic" 2 => "B. Tarlan" 3 => "F. Soylemezoglu" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.5301/ejo.5000555" "Revista" => array:6 [ "tituloSerie" => "Eur J Ophthalmol" "fecha" => "2015" "volumen" => "25" "paginaInicial" => "288" "paginaFinal" => "292" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25612656" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0065" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Orbital xanthogranuloma in an adult patient with xanthelasma palpebrarum and hypercholesterolemia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "T.J. Rubinstein" 1 => "M.P. Mehta" 2 => "L. Schoenfield" 3 => "J.D. Perry" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:4 [ "tituloSerie" => "Ophthal Plast Reconstr Surg" "fecha" => "2014" "volumen" => "30" "paginaInicial" => "6" ] ] ] ] ] ] 6 => array:3 [ "identificador" => "bib0070" "etiqueta" => "7" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The clinical spectrum of xanthomatous lesions of the eyelids" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "C. Baykal" 1 => "A. Polat Ekinci" 2 => "K.D. Yazganoglu" 3 => "N. Buyukbabani" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/ijd.13637" "Revista" => array:6 [ "tituloSerie" => "Int J Dermatol" "fecha" => "2017" "volumen" => "56" "paginaInicial" => "981" "paginaFinal" => "992" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28500693" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735794/0000009400000005/v1_201904290704/S2173579419300349/v1_201904290704/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/21735794/0000009400000005/v1_201904290704/S2173579419300349/v1_201904290704/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579419300349?idApp=UINPBA00004N" ]
Journal Information
Share
Download PDF
More article options
Short communication
Adult orbital xanthogranuloma as a cause of recurrent palpebral swelling
Xantogranuloma orbitario del adulto como causa de inflamación palpebral recidivante