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WEBINO syndrome (wall-eyed bilateral internuclear ophthalmoplegia) secondary to ischemic stroke, about a case
Síndrome de WEBINO (wall-eyed bilateral internuclear ophthalmoplegia) secundario a ictus isquémico, a propósito de un caso
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(wall-eyed bilateral internuclear ophthalmoplegia) secondary to ischemic stroke, about a case" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "205" "paginaFinal" => "208" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Daniel Vázquez-Justes, Andrés Martín-Cucó, Yhovany Gallego-Sánchez, Mikel Vicente-Pascual" "autores" => array:4 [ 0 => array:3 [ "nombre" => "Daniel" "apellidos" => "Vázquez-Justes" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "Andrés" "apellidos" => "Martín-Cucó" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Yhovany" "apellidos" => "Gallego-Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:4 [ "nombre" => "Mikel" "apellidos" => "Vicente-Pascual" "email" => array:1 [ 0 => "mvpascual.1@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" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Universitari Arnau de Vilanova, Lleida, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Centro Atención Primaria Bordeta-Magraners, Lleida, 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" => "Síndrome de WEBINO (wall-eyed bilateral internuclear ophthalmoplegia) secundario a ictus isquémico, a propósito de un caso" ] ] "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" => 551 "Ancho" => 1500 "Tamanyo" => 306642 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Primary gaze position. Photo taken 2 weeks after release. Left photo: right eye exotropia; right photo: left eye exotropia.</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 WEBINO syndrome (wall-eyed bilateral internuclear ophthalmoplegia) is a clinic syndrome characterized by alterations of extrinsic ocular motility, expressed as a bilateral deficit in ocular adduction, bilateral horizontal nystagmus to abduction, as well as the presence of exotropia in primary gaze. The WEBINO term was first utilized in 1971.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The physiopathology of this syndrome involves compromise of both longitudinal medial fascicles, superior mesencephalic tegmentum and possible compromise of subnuclei of oculomotor nerves the coordination of which is crucial for adequate execution of horizontal conjugate gaze.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinic case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">Male, 68 years of age, who suddenly experienced dizziness, diplopia and gait instability. Clinic history included liver transplant due to cirrhosis, DM type II and dyslipidemia. He had cholecystectomy due to repetition cholangitis. Active treatments include Advagraf® 2<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h (tacrolimus, Astellas Pharma Europe, Ireland), Pantoprazol TEVAGEN 20<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h (TEVA PHARMA, Zaragoza, Spain) and Daonil® 5<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h (Glibenclamide) (SANOFI, Madrid, Spain).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Neurological examination showed large angle exotropia and bilateral internuclear ophthalmoplegia (LEN) which rendered convergence impossible, as well as the presence of horizontal nystagmus to abduction in both eyes. No oblique vertical deviations were found. Superior vertical gaze was also limited with evoked vertical nystagmus with gaze in supraversion. Inferior vertical gaze was not limited. Intrinsic ocular motility did not exhibit alterations. Visual acuity was preserved and ocular fundus examination was normal. Gait was highly unstable with significant ataxia and inability to walk unassisted.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Cranial MR reveals the presence of a lesion with restriction in diffusion sequences (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), compatible with acute ischemic infarct in the middle line of the mesencephalic tegmentum, bilaterally involving the medial longitudinal fascicle as well as the pretectal area.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">During admission, the patient experienced remarkable improvement in diplopia as well as in gait stability, but even so significant functional limitation subsisted on the date of release, requiring a walker. Treatment consisted in anti-aggregating therapy with Clopidogrel STADA 75<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h (STADA, Barcelona, Spain) and alternating occlusion of both eyes. Two weeks after release, the patient was assessed in outpatient neurology practices showing considerable clinic improvement, independent gait and autonomy for basic daily life activities. Despite significant improvements, slight alternating exotropia subsisted in primary gaze (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) (Appendix B video).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">The WEBINO syndrome is an infrequent syndrome consisting in primary gaze exotropia, bilateral deficit for ocular adduction with the impossibility of convergence as well as horizontal nystagmus in the outer horizontal gaze of both eyes. The anatomic substrate of said syndrome is subject to debate although the most accepted hypothesis proposes bilateral compromise of the medial longitudinal fascicle as well as of subnuclei of medial rectus, which explains primary gaze exotropia. Suspicion about this entity involves differential diagnostic with the one-and-a-half syndrome, in which one of the eyes exhibits complete horizontal gaze paresis while the other exhibits preserved abduction.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The anatomopathological substrate is different and consists in a lesion of the pontine paramedian reticular formation as well as the ipsilateral medial longitudinal fascicle. In addition, bilateral LEN should be considered, which would involve a lesion of the bilateral medial longitudinal fascicle but without primary gaze exotropia.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The anatomopathological substrate of the WEBINO syndrome extends to other related regions. In the present patient, it compromised the pretectal area, which would explain the ataxic gait. Similar cases have been described of patients<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> with documented superior vertical gaze compromise. The origin of said alteration remains under discussion. It has been proposed that said compromise could be due to the concomitant compromise of the rostral nucleus of the medial longitudinal fascicle due to the interruption of the oculovestibular pathways.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">WEBINO syndrome etiologies most frequently include ischemia or demyelinization. Other less frequent etiologies include infection, trauma or iatrogenia after surgery. In addition, pharmacological iatrogenia was considered in the present patient due to the fact that reversible bilateral LEN cases have been described related to treatment with tacrolimus.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> However, in contrast with the present patient, said cases exhibited no visible lesions in the cranial MR. Tacrolimus levels within normal ranges as well as the presence of a lesion with diffusion restriction in MR confirmed the ischemic etiology.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Functional prognosis has not been entirely established. Morbimortality depends on the underlying etiology and treatment possibilities. Some studies have documented a more favorable visual recovery prognosis when ophthalmoplegia was isolated and the patients did not exhibit additional neurological compromise signs during examination.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> A review of patients with ischemic LEN exhibiting additional neurological compromise and visual recovery was established in 38% of cases.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> In the present case, despite significant baseline ataxia associated to diplopia, the patient exhibited a significant recovery in gait functionality, recovering the ability to walk with the aid of a walking stick as well as improvement of diplopia 2 weeks since onset.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Multiple treatment are available, from conservative such as alternating occlusion of both eyes up to aggressive treatments that include surgery. Botulinum toxin injections constitute a further option.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In the present case, due to the significant improvements and functional independence after 2 weeks, it was decided to continue with conservative treatment consisting in alternating occlusion of both eyes, together with secondary prevention with oral antiaggregants, antihypertensives and rehabilitation.</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion, even though the WEBINO syndrome is an infrequent occurrence, being aware of its existence facilitates the determination of the mesencephalic anatomical location, which helps to indicate supplementary examinations to determine its etiology. Differential diagnostic includes bilateral LEN and the one-and-a-half syndrome. In addition to addressing the main disease, treatment options include alternating occlusion of both eyes, botulinum toxin injections or surgery. Prognosis and recovery expectations should be based on the underlying etiology but are not well established. Additional research is required in this regard.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interests</span><p id="par0055" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1328702" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1224534" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1328703" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1224535" "titulo" => "Palabras clave" ] 4 => array:3 [ "identificador" => "sec0005" "titulo" => "Introduction" "secciones" => array:1 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case report" ] ] ] 5 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 6 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interests" ] 7 => array:2 [ "identificador" => "xack458759" "titulo" => "Acknowledgments" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-11-06" "fechaAceptado" => "2019-12-17" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1224534" "palabras" => array:6 [ 0 => "WEBINO syndrome" 1 => "Internuclear ophthalmoplegia" 2 => "Ischemic stroke" 3 => "Midbrain" 4 => "Longitudinal medial fascicle" 5 => "Prectal area." ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1224535" "palabras" => array:6 [ 0 => "Síndrome de WEBINO" 1 => "Oftalmoplejia internuclear" 2 => "Ictus isquémico" 3 => "Mesencéfalo" 4 => "Fascículo longitudinal medial" 5 => "Área pretectal." ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">WEBINO syndrome is characterized by bilateral adduction impairment, nystagmus of the abducting eye, and primary gaze exotropia. We present the case of a 68-year-old man who was initially attended in emergency department with sudden onset diplopia. Neurological exploration revealed WEBINO and gait ataxia. Relevant medical history included liver transplantation and subsequent tacrolimus prescription. Complementary exams revealed ischemic lesion in mesencephalic tegmentum, involving medial longitudinal fasciculus and prectal area. WEBINO syndrome is unfrequent. Among its etiologies, ischemic and demyelinating are the most frequent. In our case, iatrogenic etiology was also considered. Clinical recognition of this syndrome is required to perform adequate exams in order to reach diagnosis.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El síndrome de WEBINO se caracteriza por un déficit bilateral en la aducción ocular, nistagmo horizontal bilateral a la abducción, así como la presencia de exotropia en la mirada primaria. Presentamos el caso de un paciente varón de 68 años, que se presentó en el servicio de urgencias con diplopia de inicio súbito. La exploración fue compatible con síndrome de WEBINO, así como ataxia de la marcha. Entre los antecedentes relevantes, trasplantado hepático en tratamiento con tacrólimus. Las exploraciones complementarias pusieron de manifiesto la existencia de una lesión isquémica en tegmento mesencefálico, involucrando el fascículo longitudinal medial de forma bilateral así como el área pretectal. El síndrome de WEBINO es un síndrome poco frecuente. Entre las etiologías destacan la isquémica y la desmielinizante como las más frecuentes. En nuestro caso también se consideró la iatrogenia. La lesión asienta en un área anatómica específica, siendo clave su conocimiento para la realización de exploraciones complementarias que lleven al diagnóstico etiológico específico.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Vázquez-Justes D., Martín-Cucó A., Gallego-Sánchez Y., Vicente-Pascual M. Síndrome de WEBINO (wall-eyed bilateral internuclear ophthalmoplegia) secundario a ictus isquémico, a propósito de un caso. Arch Soc Esp Oftalmol. 2020;95:205–208.</p>" ] ] "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" => 1126 "Ancho" => 1500 "Tamanyo" => 120691 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cerebral magnetic resonance (DWI in the left side; FLAIR in the right side) showing acute ischemic lesion in the bilateral medial longitudinal fascicle, with extension to adjacent pretectal areas.</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" => 551 "Ancho" => 1500 "Tamanyo" => 306642 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Primary gaze position. Photo taken 2 weeks after release. Left photo: right eye exotropia; right photo: left eye exotropia.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Suplanuclear disorders of ocular control systems in man: Clinical, anatomical and physiological correlations" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "R.B. Daroff" 1 => "W.F. 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The authors wish to express their sincere gratitude to the patient because this article would not have been possible without his cooperation.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/21735794/0000009500000004/v1_202004240656/S2173579420300372/v1_202004240656/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/21735794/0000009500000004/v1_202004240656/S2173579420300372/v1_202004240656/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173579420300372?idApp=UINPBA00004N" ]
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