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Detecting progression of acute zonal occult outer retinopathy (AZOOR) with optical coherence tomography angiography: A case report
Detección de la progresión de la retinopatía externa oculta zonal aguda (AZOOR) con angiografía por tomografía de coherencia óptica: reporte de un caso
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La OCT-A <span class="elsevierStyleItalic">en face</span> reveló puntos hiperreflectantes a nivel de la zona elipsoide sobre un fondo hiporreflectante en AO (e y f), aunque más evidentes en el ojo izquierdo (OI), que corresponden a depósitos drusenoides subretinianos (g-j). Cuando se produjo la progresión (k-t), aparecieron nuevas manchas de espesor seudoincrementado (k y l) y se desarrollaron puntos hiperreflectantes más numerosos a pesar de que el fondo hiporreflectante había disminuido de tamaño en la OCT-A <span class="elsevierStyleItalic">en face</span> en AO (o y p). También hay un aumento de la penetración de la señal luminosa debido a la atrofia del epitelio pigmentario de la retina y del coriocapilar (q-t).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "C. de los Santos, J.M. Herreras, L. Cocho" "autores" => array:3 [ 0 => array:2 [ "nombre" => "C." "apellidos" => "de los Santos" ] 1 => array:2 [ "nombre" => "J.M." 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It is associated with perilesional edema, enhances homogeneously and extends towards the left orbit, middle fossa and temporalis muscle (asterisks).</p> <p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">C and D) Coronal slices of the pre-surgical cranial MRI where, in addition to the mass visible in the CT scan, another lesion with extra-axial characteristics and plaque morphology can be seen in the right frontal convexity (arrow in C). After Gadolinium administration, both lesions showed homogeneous enhancement (C). The T2-weighted sequence shows vasogenic edema around hypointense lesions.</p> <p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">E and F) Axial (E) and coronal (F) MRI slices (T1-weighted sequence) after contrast administration, obtained after cancer treatment. There is a large reduction in the volume of the lesion but with persistent diffuse involvement of the left sphenoid and temporal bone.</p> <p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">G and H) Cranial CT scan after intravenous contrast administration, performed during the patient's follow-up, showing left frontotemporal postoperative malignancy (asterisk), with no focal lesions suggestive of tumour persistence or local recurrence.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "E. Barrero Ruíz, R. Carrasco Moro" "autores" => array:2 [ 0 => array:2 [ "nombre" => "E." "apellidos" => "Barrero Ruíz" ] 1 => array:2 [ "nombre" => "R." 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Barrera, A. Bustamante, M. Marín-Cornuy, P. Aguila-Torres" "autores" => array:4 [ 0 => array:2 [ "nombre" => "B." "apellidos" => "Barrera" ] 1 => array:2 [ "nombre" => "A." "apellidos" => "Bustamante" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Marín-Cornuy" ] 3 => array:2 [ "nombre" => "P." "apellidos" => "Aguila-Torres" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0365669123001521" "doi" => "10.1016/j.oftal.2023.07.011" "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/S0365669123001521?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S217357942300138X?idApp=UINPBA00004N" "url" => "/21735794/0000009800000010/v2_202311091359/S217357942300138X/v2_202311091359/en/main.assets" ] "en" => array:19 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Short communication</span>" "titulo" => "Detecting progression of acute zonal occult outer retinopathy (AZOOR) with optical coherence tomography angiography: A case report" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "595" "paginaFinal" => "600" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "C. de los Santos, J.M. Herreras, L. Cocho" "autores" => array:3 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "de los Santos" "email" => array:1 [ 0 => "hes.oftalmologia.cs@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "J.M." "apellidos" => "Herreras" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "L." "apellidos" => "Cocho" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "IOBA (Instituto de Oftalmobiología Aplicada), Universidad de Valladolid, Valladolid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento de Oftalmología, Hospital Clínico Universitario de Valladolid, Valladolid, 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" => "Detección de la progresión de la retinopatía externa oculta zonal aguda (AZOOR) con angiografía por tomografía de coherencia óptica: reporte de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1660 "Ancho" => 3341 "Tamanyo" => 556624 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Optical coherence tomography angiography (OCT-A) at first visit showing reduced thickness (a and b) and increased decorrelation signal at the deeper capillary plexus along with projection artifacts of the superficial capillary plexus (c and d) in both eyes (BE) due to outer retina atrophy (g–j). En Face OCT-A revealed hyperreflective dots at ellipsoid zone level over a hyporreflective background in BE (e and f), although more evident in left eye (LE), which correspond to subretinal drusenoid deposits (g–j). When progression occurred (k–t), new spots of pseudo-increased thickness appeared (k and l) and more numerous hyperreflective dots developed despite the hyporeflective background had decreased in size on En Face OCT-A BE (o and p). There is also increased penetration of light signal due to retinal pigment epithelium and choriocapillaris atrophy (q–t).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Acute zonal occult outer retinopathy (AZOOR) is a rare primary disorder of the outer retina (photoreceptoritis) first described by Gass in 1992<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> affecting predominantly young and middle-aged women (13–63 years) with myopia,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Patients often complain of acute and unilateral onset, although ¾ progresses to bilateral,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> of loss of function of one or more zones of the outer retina (photopsia and scotomas), sometimes accompanied by photophobia, mimicking migraine, and decreased contrast sensitivity. Fundoscopic changes are absent or minimal at the beginning, however, alterations may be evident in the electroretinogram (ERG) with subsequent visual field defects. Multimodal imaging by fundus autofluorescence (FAF), optical coherence tomography (OCT), and indocyanine green (ICG) angiography may show a characteristic trizonal pattern in the subacute and chronic stage.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Lesions usually stabilize in 4–6 months, and central vision remains good in most cases, although recurrences can occur in one third of cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">There are few reports using OCT angiography (OCT-A),<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5–7</span></a> nonetheless, we highlight its usefulness in detecting disease progression by presenting a case with 4-year follow-up. Furthermore, treatment is controversial given the fact some cases refer viral prodromes or history of autoimmune diseases,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and the pathogenesis has not yet been proven,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> however we report a new effective treatment option, particularly when recurrence ensues.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Clinical case</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 48-year-old woman without history of migraine complaining of superotemporal scotomas accompanied by photopsia and photophobia of 3 days of evolution in both eyes (BE), with apparent normal ocular fundus (OF), a Humphrey visual field (HVF) test reporting probable upper bitemporal quadrantanopia, a magnetic resonance imaging of the orbit and brain with contrast without retrobulbar, chiasmatic or parenchymal lesion, and normal visual evoked potentials for BE, who was referred to our institution 2 months later because of persistent symptoms. Autoimmune diseases or any recent viral disease were denied and HVF was repeated (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">At first visit, clinical examination was not remarkable including visual acuity, intraocular pressure, and anterior segment in BE, except for the OF that showed a zone of subtle depigmentation inferotemporal to the optic disc in right eye (RE) and in the peripapillary region in left eye (LE) (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Fluorescein angiography demonstrated early and late hyperfluorescence due to retinal pigment epithelium (RPE) window defect and slight optic disc staining in BE without signs of vasculitis or macular edema (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). On FAF these lesions had diffuse hyperautofluorescence demarcated by a continuous line of greater hyperautofluorescence and in later visits they became hypoautofluorescent (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A conventional electroretinogram was performed revealing mixed cone and rod disfunction and a decreased response of the Pattern ERG (PERG) and oscillatory potentials (OPs) in LE, and the multifocal ERG (MERG) showed reduced response in BE corresponding to HVF defects (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">OCT demonstrated retinal thinning due to outer retinal atrophy with loss of outer plexiform layer, outer nuclear layer, external limiting membrane, ellipsoid zone (EZ), myoid zone and interdigitation zone, and subsequent RPE and choriocapillaris (CC) atrophy in BE (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). In addition, hyperreflective subretinal deposits were seen within and on the edge of the lesions. OCT-A showed increased decorrelation in the deep plexus with projection artifacts from the superficial plexus and the En Face OCT-A at the EZ level demonstrated hyperreflective dots over a hyporeflective area that was greater than FAF changes, with additional hyporreflective lesions temporally to the main lesion in BE.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">An extensive work-up for infectious, inflammatory or neoplastic disease was performed including Treponema pallidum, Herpes simplex virus type 1 and 2, Toxocara canis, Bartonella henselae, QuantiFERON-TB Gold, serum calcium, angiotensin-converting enzyme, erythrocyte sedimentation rate, c-protein reactive, rheumatoid factor, serum complement C3 and C4, IgM and IgG anticardiolipin antibodies, antinuclear antibodies, antineutrophil cytoplasmic antibodies, tumor markers (carcinoembryonic antigen and CA 19-9), anti-titin, anti-recoverin, anti-CV2, anti-amphiphysin, anti-Ri, anti-neuronal (Ma2), anti-Yo, anti-Hu, anti-SOX1, anti-alpha enolase, anti-carbonic anhydrase antibodies and a positron emission tomography (PET) which were all negative, therefore, AZOOR was diagnosed.</p><p id="par0035" class="elsevierStylePara elsevierViewall">After worsening detected by HVF and ERG in BE at the third month of follow-up (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), immunomodulatory treatment with subcutaneous adalimumab 40 mg every 2 weeks was initiated showing bilateral improvement, nevertheless, 19 months later the patient reported an increase in symptoms and progression was observed in HVF, ERG and OCT-A in BE (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 3</a>), therefore, adalimumab and anti-adalimumab antibodies levels in serum were tested reporting normal [9.4 μg/mL (reference 5–12) and <10 ng/mL (reference <10 ng/mL) respectively], however, mycophenolate mofetil was added 2 g/day with later improvement in BE. Currently, with 4 years of follow-up, the patient reports being asymptomatic, maintains a visual acuity of 1.0 in both eyes, and stability is recorded both in HVF, ERG and multimodal imaging.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">AZOOR is a primary photoreceptoritis with secondary involvement of the RPE and CC. At the beginning it may not be clinically apparent, nevertheless a grayish-white demarcation line may be seen occasionally between the affected and the normal retina if there is active expansion of the disease, which disappears within weeks and is replaced with an orange area. When it progresses, it can present pigment deposition with zonal or multizonal retinochoroidal atrophy, often seen as a peripapillary depigmentation, and in advanced stages is associated with focal perivenous sheathing and retinal arteriolar attenuation, mimicking sectoral or asymmetric retinitis pigmentosa, syphilis, autoimmune retinopathy, diffuse unilateral subacute neuroretinitis and retinal vasculitis.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a> nevertheless, these entities were ruled out in this patient.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The electroretinogram may exhibit disfunction of both cones and rods as well as reduced amplitude in OPs, suggesting that inner retina may be secondarily affected,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> since photoreceptors provide electrical input signals to the more proximal cells that generate the Ops. HVF test may exhibit scotomas that suggest retrobulbar optic neuritis or intracranial tumor which were ruled out in this patient.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Multimodal imaging by FAF, OCT and ICG angiography may show a characteristic trizonal pattern<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> consisting of normal retina (zone 1), transition line of outer retina atrophy (zone 2), and an area with additional RPE and CC atrophy (zone 3). Paraneoplastic and nonparaneoplastic autoimmune retinopathy may also show outer retinal dysfunction resembling AZOOR, but despite the absence of systemic symptoms, antiretinal antibodies, tumor markers and PET-Scan were negative confirming the diagnosis. Several reports have detected OCT-A changes in AZOOR which include increase in deep flow density as a possible source of mediators of inflammation in contrast to inactive phase where it is decreased,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> although this finding was not seen in our patient. Furthermore, others have reported increased decorrelation signal at the level of the deep capillary plexus with projection artifacts from the superficial capillary plexus<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> and increased hyperreflective dots on En Face OCT-A at EZ level<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> as in our patient, the latter suggesting outer segments of degraded photoreceptors, however, to the best of our knowledge, this is the first case report to address progression of disease in a long-term context and correlate this findings with worsening in the ERG and HVF.</p><p id="par0055" class="elsevierStylePara elsevierViewall">There is no consensus on whether treatment alters or not the natural history of the disease because of limited reports in the literature, and the use of antivirals such as acyclovir and valacyclovir is anecdotal with an efficacy not yet proven in this pathology. Moreover, the response to systemic corticosteroids and/or immunosuppressants such as methotrexate, mycophenolate mofetil, azathioprine, cyclosporine, infliximab, and adalimumab is variable. Nevertheless, this patient had a good clinical response combining adalimumab and mycophenolate mofetil when recurred, and so far, there is no more evidence in the literature of this therapeutic approach.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In conclusion, multimodal imaging is essential to distinguish it from other diseases, however, since this is a primary photoreceptoritis, En Face OCTA at EZ level seems to be a potential imaging technique to monitor progression and treatment response along with ERG, HVF and FAF. Additionally, the combination of adalimumab with mycophenolate mofetil is good treatment option in recurrent disease.</p></span><span id="sec1065" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect1125">Funding</span><p id="par1320" class="elsevierStylePara elsevierViewall">The authors confirm that they have not received funding for this article.the realisation of this article.</p></span><span id="sec0070" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0130">Conflict of interest</span><p id="par1325" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres2007162" "titulo" => "Abstract" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1719761" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres2007163" "titulo" => "Resumen" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1719762" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec1065" "titulo" => "Funding" ] 8 => array:2 [ "identificador" => "sec0070" "titulo" => "Conflict of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2023-03-14" "fechaAceptado" => "2023-05-30" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1719761" "palabras" => array:4 [ 0 => "Acute zonal occult outer retinopathy" 1 => "Adalimumab" 2 => "Mycophenolate mofetil" 3 => "Optic coherence tomography angiography" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1719762" "palabras" => array:4 [ 0 => "Retinopatía externa oculta zonal aguda" 1 => "Adalimumab" 2 => "Micofenolato de mofetilo" 3 => "Angiografía por 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">Clinical case</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">A 48-year-old woman with persistent superotemporal scotomas and photopsias for 2 months, and depigmented zones in the retina of both eyes with a trizonal pattern on multimodal imaging. Brain magnetic resonance imaging, positron emission tomography, antiretinal antibodies, immunological, infectious and tumor markers tests were negative, thus acute zonal occult outer retinopathy was diagnosed. Patient was treated with adalimumab. Nevertheless, 19 months later symptoms increased, and progression was detected on optic coherence tomography angiography, as well as in Humphrey visual field test and electroretinogram, thus, mycophenolate mofetil was added showing improvement and stabilization of the disease in a 4-year follow-up.</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Optic coherence tomography angiography may be a potential tool to monitor progression and response to treatment in addition to other imaging modalities in acute zonal occult outer retinopathy, and the combination of adalimumab and mycophenolate may be useful in recurrent disease.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] "es" => array:3 [ "titulo" => "Resumen" "resumen" => "<span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Caso clínico</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Mujer de 48 años con escotomas superotemporales persistentes y fotopsias de 2 meses de evolución y zonas despigmentadas en retina de ambos ojos con patrón trizonal en imagen multimodal. La resonancia magnética cerebral, la tomografía por emisión de positrones, los anticuerpos antirretinianos, los marcadores inmunológicos, infecciosos y tumorales fueron negativos, por lo que se diagnosticó retinopatía externa oculta zonal aguda. La paciente fue tratada con adalimumab, sin embargo, 19 meses después los síntomas aumentaron y se detectó progresión en la angiografía por tomografía de coherencia óptica, así como también en la prueba de campo visual Humphrey y el electrorretinograma, por lo que se agregó micofenolato de mofetilo mostrando mejoría y estabilización de la enfermedad durante un seguimiento de 4 años.</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discusión</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">La angiografía por tomografía de coherencia óptica puede ser una herramienta potencial para monitorear la progresión y la respuesta al tratamiento además de otras modalidades de imagen en la retinopatía externa oculta zonal aguda, y la combinación de adalimumab y micofenolato puede ser útil en la enfermedad recurrente.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 3899 "Ancho" => 3341 "Tamanyo" => 1514906 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Humphrey visual field test at first visit showed a superior arcuate scotoma (a) in right eye (RE) and increased blind spot with paracentral extension (b) in left eye (LE) which improved after treatment in next visits in both eyes (BE) (c and d), nevertheless, a worsening was detected 19 months later in BE (e and f), so that treatment was modified showing improvement in the last visit (g and h) in BE. Electroretinogram (ERG) at first visit revealed a reduced response in the amplitude of a and b waves in rod (scotopic) and cone (flicker-30 Hz) responses (i-k), in P50 of the pattern ERG (PERG) (l) and in the oscillatory potentials (posc) (m) in LE. Multifocal ERG (MERG) exhibited a decrease in the amplitude of P1 wave in the upper macula in BE (n and o), although more accentuated in LE, which improved after treatment (p and q), but worsening was seen at 19th month (r and s).</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 2786 "Ancho" => 3341 "Tamanyo" => 533193 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Ocular fundus at first visit showing a depigmented zone due to retinochoroidal atrophy inferotemporal to the optic disc in right eye (RE) (a) and in the peripapillary region in left eye (LE) (b). At 7th month (c and d), 19th month (e and f), and four years later (g and h) lesions became more evident with pigment deposition and an additional nasal zone was clinically apparent in LE in the last visit (h). On fundus autofluorescence lesions showed diffuse hyperautofluorescence in both eyes (BE) demarcated by a continuous line of greater hyperautofluorescence (i and j) and two smaller lesions were noticeable nasally in LE. At 7th month (k and l) there was stippling hypoautofluorescence in the center of the lesions that became more regular at 19th month (m and n) and four years later (o and p). Fluorescein angiography at first visit showed late hyperfluorescence due to retinal pigmentary epithelium window defect with mild optic disc staining and slight arteriolar attenuation in affected areas of BE (q and r).</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1660 "Ancho" => 3341 "Tamanyo" => 556624 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Optical coherence tomography angiography (OCT-A) at first visit showing reduced thickness (a and b) and increased decorrelation signal at the deeper capillary plexus along with projection artifacts of the superficial capillary plexus (c and d) in both eyes (BE) due to outer retina atrophy (g–j). En Face OCT-A revealed hyperreflective dots at ellipsoid zone level over a hyporreflective background in BE (e and f), although more evident in left eye (LE), which correspond to subretinal drusenoid deposits (g–j). When progression occurred (k–t), new spots of pseudo-increased thickness appeared (k and l) and more numerous hyperreflective dots developed despite the hyporeflective background had decreased in size on En Face OCT-A BE (o and p). There is also increased penetration of light signal due to retinal pigment epithelium and choriocapillaris atrophy (q–t).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Acute zonal occult outer retinopathy. 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