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Bilateral acute iris depigmentation and bilateral acute iris transillumination syndrome
Síndrome de despigmentación aguda bilateral de iris y transiluminación aguda bilateral de iris
T. Rueda-Ruedaa, L.J. Sánchez-Vicenteb, A. Moruno-Rodríguezc,
, J. Monge-Esquivelc, A. Muñoz-Moralesd, F. López-Herreroe
Corresponding author
a Servicio de Oftalmología, Sección de Uveítis, Hospital Universitario Virgen del Rocío, Sevilla, Spain
b Servicio de Oftalmología, Sección de Retina Quirúrgica, Hospital Universitario Virgen del Rocío, Sevilla, Spain
c Servicio de Oftalmología, Sección General, Hospital Universitario Virgen del Rocío, Sevilla, Spain
d Servicio de Oftalmología, Sección de Córnea, Hospital Universitario Virgen del Rocío, Sevilla, Spain
e Servicio de Oftalmología, Sección de Retina Médica, Hospital Universitario Virgen del Rocío, Sevilla, Spain
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"apellidos" => "López-Herrero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0025" ] ] ] ] "afiliaciones" => array:5 [ 0 => array:3 [ "entidad" => "Servicio de Oftalmología, Sección de Uveítis, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Oftalmología, Sección de Retina Quirúrgica, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Oftalmología, Sección General, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Oftalmología, Sección de Córnea, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] 4 => array:3 [ "entidad" => "Servicio de Oftalmología, Sección de Retina Médica, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "e" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de despigmentación aguda bilateral de iris y transiluminación aguda bilateral de iris" ] ] "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" => 274 "Ancho" => 1250 "Tamanyo" => 23330 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Anterior segment optical coherence tomography showing iris location in relation to the lens, discarding the existence of anterior iris concavity and therefore the possibility of pigment dispersion syndrome.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">The bilateral acute transillumination of iris syndrome (BATI) was first described in 2011 by Tugal-Tutkun et al. who reported 26 patients with acute and bilateral clinic characterized by the loss of iridian pigment, iris transillumination, pigment dispersion in anterior chamber, pupils in medium mydriasis with poor or null response to light and increased intraocular pressure (IOP) in some cases.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">In 2006 the same author described the bilateral acute depigmentation of the iris syndrome (BADI), with pigment dispersion in anterior chamber, iris stroma depigmentation and pigment deposits in anterior chamber.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">BADI exhibits a more benign course with absence of transillumination, considerably less frequent IOP increases and absence of pupil compromise.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The case of a patient with BATI is presented, exhibiting iris stromal depigmentation areas which are typical of said syndrome. This supports the idea that both entities are parts of the same disease.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinic case report</span><p id="par0025" class="elsevierStylePara elsevierViewall">Patient, 34, referred from the Emergency Dept. due to acute bilateral panuveitis with ocular hypertension. The patient referred sudden loss of vision in both eyes with maximum corrected visual acuity (MCVA) of 0.2 in the right eye (RE) and 0.3 in the left eye (LE). IOP was 32<span class="elsevierStyleHsp" style=""></span>mmHg in RE and 42<span class="elsevierStyleHsp" style=""></span>mmHg in LE. The episode was preceded by fever with headaches related to tonsillitis treated with azithromycin and paracetamol. The condition was treated with oral and topical corticoids (prednisolone at 3<span class="elsevierStyleHsp" style=""></span>h intervals) and topical hypotensors (0.5% brinzolamide, brimonidine and timolol every 12<span class="elsevierStyleHsp" style=""></span>h).</p><p id="par0030" class="elsevierStylePara elsevierViewall">Examined at the practice 2 days later, the patient exhibited significant improvement of symptoms. MCVA had improved to 0.6 in the RE and 0.5 in the LE, while IOP had diminished to 20 and 14<span class="elsevierStyleHsp" style=""></span>mmHg in the RE and LE, respectively. Biomicroscopy showed hardly reactive medium mydriasis. The iris showed depigmentation areas with some transillumination areas. Anterior chamber evidenced the existence of pigment dispersion with pigment deposits in the corneal endothelium and anterior chamber angle (visible with gonioscopy). No inflammatory keratic precipitates were observed (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). Similarly, iris concave surface or posterior displacement was not observed. These findings were confirmed with anterior segment optical coherence tomography (DRI OCT Triton plus, Topcon Medical Systems, Inc.), discarding the possibility of pigment dispersion syndrome (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">No signs of inflammation were observed in the vitreous or the retina. Papilla exhibited poorly defined edges, initially attributed to the possible presence of drusen in the optic disc. This was subsequently confirmed with OCT and ocular echography (Aviso, Quantel MedicaLikewise, the presence of macular edema was discarded by means of macular OCT.</p><p id="par0040" class="elsevierStylePara elsevierViewall">In the absence of inflammatory cells, posterior synechiae and keratic precipitates that could point to iridocyclitis, a suspected diagnostic of BADI transillumination was established, initiating uveitis study to discard other causes. Analytics and serological studies produced normal results (<span class="elsevierStyleItalic">Bartonella</span>, <span class="elsevierStyleItalic">Coxiella</span>, <span class="elsevierStyleItalic">Rickettsia</span>, cytomegalovirus, Epstein–Barr virus, hepatitis B and <span class="elsevierStyleSmallCaps">C</span> virus, herpes zoster, herpes simplex, <span class="elsevierStyleItalic">Toxocara</span>, toxoplasma and syphilis).</p><p id="par0045" class="elsevierStylePara elsevierViewall">Six months later, MCVA was 1.0 in both eyes with IOP under control with 0.5% brinzolamide, brimonidine and timolol every 12<span class="elsevierStyleHsp" style=""></span>h.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">BADI as well as BATI exhibit typical characteristics enabling differentiation between both. In the case of BADI, geographic or diffuse depigmentation of the iridium stroma is found, without papillary compromise and absence of transillumination defects. In contrast, patients with BATI presented with dilated and atonic pupils, transillumination defects and absence of iris stroma compromise. In BADI, pigment dispersion occurs from the iris stroma while in BATI it arises from the pigment epithelium of the iris. In addition, BADI exhibits a more benign course with lower ocular hypertension incidence which is generally temporary as well as reversibility of iris alterations in some cases.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Despite the above differences, some authors consider the possibility that both entities constitute different expressions of the same disease, basically due to the presence of common characteristics such as debut with acute red eye together with photophobia and pigment dispersion, higher prevalence in females, the fact of being preceded by high airways infection and positive response to corticoids.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The present patient exhibited geographic depigmentation of the iris associated to typical BATI characteristics such as iris transillumination, dilated and atonic pupils and increased IOP. The coexistence of said findings in the same patient gives rise to the possibility of an intermediate clinic in evolution between BADI and BATI, supporting the idea that both entities are expressions of the same disease. Accordingly, BADI would be an earlier expression with less severe clinic expressions than BATI, which would be found in the other extreme of the disease, with severe signs and symptoms.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The etiology of the disease is unknown. Some authors have related it to the use of moxifloxacin and other quinolones<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4,5</span></a> or other antibiotics.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> Other authors<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> do not endorse said hypothesis. Active viral infection has also been suggested, including herpes.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2,7</span></a> On the basis of a possible viral cause, topical antivirals have been utilized (0.15% ganciclovir ophthalmic gel) in some isolated cases with positive response to treatment.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> However, Tugal-Tutkun et al. consider that the reversibility of changes in the iris is not compatible with active viral inflammation or infection. Said author argues that reversibility could be explained by a neuropathic effect.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Amin et al. reported a simultaneous occurrence in 2 twins. On the basis of this finding and the fact that the majority of cases have been described in Turkey in geographically close locations and with common genetic characteristics, said author proposed the possibility of genetic predisposition toward the disease.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">A differential diagnostic must consider the possibility of iridocyclitis, particularly due to herpes virus and cytomegalovirus which could give rise to hypotensive uveitis, Fuchs’ uveitis syndrome, pigment dispersion syndrome and pseudoexfoliative syndrome.</p><p id="par0080" class="elsevierStylePara elsevierViewall">The BADI-BATI syndrome constitutes a new clinic entity of unknown etiology, the diagnostic of which is based on a group of clinic characteristics. Both entities could be part of the range of a single disease.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interests</span><p id="par0085" class="elsevierStylePara elsevierViewall">No conflict of interests was disclosed by the authors who also state that they have not received any financial support.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1213871" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1129542" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1213872" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1129543" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interests" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-07-03" "fechaAceptado" => "2018-11-05" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1129542" "palabras" => array:6 [ 0 => "Iris transillumination" 1 => "Iris depigmentation" 2 => "Pigment dispersion" 3 => "Intraocular pressure" 4 => "Uveitis" 5 => "Masquerade syndromes" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1129543" "palabras" => array:6 [ 0 => "Transiluminación de iris" 1 => "Despigmentación de iris" 2 => "Dispersión pigmentaria" 3 => "Presión intraocular" 4 => "Uveítis" 5 => "Síndromes de enmascaramiento" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The case is presented of a 32 year-old male who arrived with acute bilateral symptoms with blurred vision, red eye, severe photophobia and severe ocular pain after suffering from a flu-like syndrome. The patient presented with a clinical picture of bilateral involvement characterized by pupils in mid-mydriasis, scarcely reactive to light, iris transillumination, diffuse depigmentation of the iridian stroma, pigment dispersion in the anterior chamber, and ocular hypertension. After the eye examination an inflammatory syndrome and pigmentary glaucoma were ruled out.</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The patient showed depigmentation characteristics as well as bilateral iris transillumination. Both conditions could form part of the spectrum of the same disease.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Varón de 32 años que acude con un cuadro agudo bilateral caracterizado por visión borrosa, ojo rojo, fotofobia severa y dolor ocular tras un cuadro seudogripal. El paciente presentaba un cuadro con afectación bilateral caracterizado por pupilas en midriasis media, escasamente reactivas a la luz, transiluminación del iris, despigmentación difusa del estroma iridiano, dispersión de pigmento en la cámara anterior e hipertensión ocular. Tras el examen ocular se descartó un cuadro inflamatorio y un glaucoma pigmentario.</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El paciente presentaba características tanto de la despigmentación como de la transiluminación bilateral de iris. Ambas entidades podrían formar parte del espectro de la misma enfermedad.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Rueda-Rueda T, Sánchez-Vicente LJ, Moruno-Rodríguez A, Monge-Esquivel J, Muñoz-Morales A, López-Herrero F. Síndrome de despigmentación aguda bilateral de iris y transiluminación aguda bilateral de iris. Arch Soc Esp Oftalmol. 2019;94:355–358.</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" => 2778 "Ancho" => 2083 "Tamanyo" => 337752 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">(a) Pupils in medium mydriasis, irregular and with depigmentation areas; (b) pigment deposits in the endothelium; (c) pigment deposits in anterior chamber angle.</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" => 1273 "Ancho" => 900 "Tamanyo" => 80149 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Small transillumination areas shown with blue arrows.</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" => 274 "Ancho" => 1250 "Tamanyo" => 23330 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Anterior segment optical coherence tomography showing iris location in relation to the lens, discarding the existence of anterior iris concavity and therefore the possibility of pigment dispersion syndrome.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Bilateral acute iris transillumination" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "I. 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