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Biopsia de OM y de <span class="elsevierStyleItalic">cavum</span>: infiltrado de células inflamatorias crónicas, pequeños focos de necrosis, degeneración fibrinoide, tejido de granulación y extravasación de células rojas (tinción hematoxilina-eosina 40×).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Bharti Tulsidas-Mahtani, Herminio Pérez-Garrigues, Noelia Muñoz-Fernández, Constantino Morera" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Bharti" "apellidos" => "Tulsidas-Mahtani" ] 1 => array:2 [ "nombre" => "Herminio" "apellidos" => "Pérez-Garrigues" ] 2 => array:2 [ "nombre" => "Noelia" "apellidos" => "Muñoz-Fernández" ] 3 => array:2 [ "nombre" => "Constantino" "apellidos" => "Morera" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S2173573514000271" "doi" => "10.1016/j.otoeng.2014.03.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573514000271?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001651912002002?idApp=UINPBA00004N" "url" => "/00016519/0000006500000002/v1_201403120114/S0001651912002002/v1_201403120114/es/main.assets" ] ] "itemSiguiente" => array:19 [ "pii" => "S2173573514000283" "issn" => "21735735" "doi" => "10.1016/j.otoeng.2012.09.010" "estado" => "S300" "fechaPublicacion" => "2014-03-01" "aid" => "433" "copyright" => "Elsevier España, S.L." "documento" => "simple-article" "crossmark" => 0 "subdocumento" => "crp" "cita" => "Acta Otorrinolaringol Esp. 2014;65:133-4" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:2 [ "total" => 1773 "formatos" => array:3 [ "EPUB" => 50 "HTML" => 1142 "PDF" => 581 ] ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Study</span>" "titulo" => "Clear Cell Carcinoma of the Base of the Tongue" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "133" "paginaFinal" => "134" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Carcinoma de células claras en la base de la lengua" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 864 "Ancho" => 999 "Tamanyo" => 120921 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">NMR, sagittal cut: mass of 4.4<span class="elsevierStyleHsp" style=""></span>cm×4.5<span class="elsevierStyleHsp" style=""></span>cm×2.6<span class="elsevierStyleHsp" style=""></span>cm in the base of the tongue, mildly aggressive, with well-defined margins and lacking areas of necrosis.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Soraya Moreno Zafra, Marcelino Rodríguez Verdugo, Rafael Hernández López" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Soraya" "apellidos" => "Moreno Zafra" ] 1 => array:2 [ "nombre" => "Marcelino" "apellidos" => "Rodríguez Verdugo" ] 2 => array:2 [ "nombre" => "Rafael" "apellidos" => "Hernández López" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001651912002038" "doi" => "10.1016/j.otorri.2012.09.004" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001651912002038?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573514000283?idApp=UINPBA00004N" "url" => "/21735735/0000006500000002/v1_201404170146/S2173573514000283/v1_201404170146/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S217357351400026X" "issn" => "21735735" "doi" => "10.1016/j.otoeng.2012.06.001" "estado" => "S300" "fechaPublicacion" => "2014-03-01" "aid" => "424" "copyright" => "Elsevier España, S.L." "documento" => "simple-article" "crossmark" => 0 "subdocumento" => "crp" "cita" => "Acta Otorrinolaringol Esp. 2014;65:129-30" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:2 [ "total" => 2924 "formatos" => array:3 [ "EPUB" => 56 "HTML" => 1717 "PDF" => 1151 ] ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case study</span>" "titulo" => "Abscess Secondary to Tophaceous Gout: A Rare Cause of Parotid Region Tumour" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "129" "paginaFinal" => "130" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Absceso secundario a tofo gotoso: una causa rara de tumoración en la región parotídea" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 942 "Ancho" => 982 "Tamanyo" => 110791 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Computed tomography image showing an increased-capture lesion with a 3-cm outer diameter in the right parotid area.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Juan Jose Góngora Lencina, Juan García-Valdecasas Bernal, Mercedes Caba Molina, Manuel Sainz Quevedo" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Juan Jose" "apellidos" => "Góngora Lencina" ] 1 => array:2 [ "nombre" => "Juan" "apellidos" => "García-Valdecasas Bernal" ] 2 => array:2 [ "nombre" => "Mercedes" "apellidos" => "Caba Molina" ] 3 => array:2 [ "nombre" => "Manuel" "apellidos" => "Sainz Quevedo" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001651912001811" "doi" => "10.1016/j.otorri.2012.06.013" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001651912001811?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S217357351400026X?idApp=UINPBA00004N" "url" => "/21735735/0000006500000002/v1_201404170146/S217357351400026X/v1_201404170146/en/main.assets" ] "en" => array:17 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Study</span>" "titulo" => "Severe Vestibulocochlear Involvement in Wegener's Granulomatosis: A Rare Presentation" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "131" "paginaFinal" => "132" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Bharti Tulsidas-Mahtani, Herminio Pérez-Garrigues, Noelia Muñoz-Fernández, Constantino Morera" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Bharti" "apellidos" => "Tulsidas-Mahtani" "email" => array:1 [ 0 => "artitop@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Herminio" "apellidos" => "Pérez-Garrigues" ] 2 => array:2 [ "nombre" => "Noelia" "apellidos" => "Muñoz-Fernández" ] 3 => array:2 [ "nombre" => "Constantino" "apellidos" => "Morera" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Otorrinolaringología, Hospital Universitario y Politécnico La Fe, Valencia, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Afectación vestibulococlear grave en granulomatosis de Wegener localizada: una presentación atípica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 770 "Ancho" => 1028 "Tamanyo" => 214978 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histopathology. Biopsy of ME and cavum: infiltrate of chronic inflammatory cells, small foci of necrosis, fibrinoid degeneration, granulation tissue and extravasation of red blood cells (hematoxylin–eosin staining, 40×).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical Case</span><p id="par0005" class="elsevierStylePara elsevierViewall">We present the clinical case of 61-year-old female, lacking relevant antecedents, whose condition began with tinnitus and hearing loss of the right ear (RE). At 4 months she presented an episode of sudden vertigo, followed by continuous dizziness and imbalance. She received a diagnosis of otitis media with effusion (OME), but did not improve after various treatments. After that, she developed hearing loss of the left ear, with a sensation of fluctuating blockage, tinnitus and right facial paresis. One year later, the hearing loss had progressed toward bilateral cophosis and the imbalance worsened. However, the facial paralysis facial subsided. <span class="elsevierStyleItalic">Otomicroscopy exploration:</span> image compatible with OME. <span class="elsevierStyleItalic">Ear and neurological examination:</span> no spontaneous nystagmus, Halmagyi test with corrective ocular with saccades on both sides, instability in Romberg and Unterberger test, absence of response to caloric stimulation (even with ice water) and in the kinetic vestibular rotating chair test (at both low and intermediate frequencies), the patient presented only minimal response to high frequency stimulation, with a gain of merely 0.12–0.64<span class="elsevierStyleHsp" style=""></span>Hz of rotation. <span class="elsevierStyleItalic">Imaging tests:</span> computed axial tomography (CAT) scan revealed occupation of the maxillary sinus and of bilateral middle ears (ME). On magnetic resonance imaging (MRI), there was thickening of the left posterior wall of the cavum and signs of bilateral damage, stronger in RE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). <span class="elsevierStyleItalic">Immunological analysis:</span> This confirmed positivity for p-ANCA type antibodies with titers of 1:64. <span class="elsevierStyleItalic">Histological analysis:</span> biopsy of ME, cavum and maxillary sinuses showed granulation tissue, small foci of necrosis, fibrinoid degeneration, inflammatory infiltrate and extravasation of red blood cells (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Treatment was initiated with prednisone at 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day and aziatropine at 100<span class="elsevierStyleHsp" style=""></span>mg/day. At 4 months the patient was clinically stable and drug treatment was suspended: The vestibular sequelae were treated with vestibular rehabilitation vestibular and the auditory ones with placement of a cochlear implant (CI) (Medel) in the left ear at 18 months after onset of clinical picture. A year later, she walks without help and presents an audiometric threshold in free field of 40<span class="elsevierStyleHsp" style=""></span>db (mean: 225–4000<span class="elsevierStyleHsp" style=""></span>Hz) with 48% of two-syllable discrimination.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0010" class="elsevierStylePara elsevierViewall">Wegener's granulomatosis (WG) is a systemic vasculitis necrotizing with granulomatous inflammation of small and medium blood vessels.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It normally has a local presentation, in which the clinical findings are limited to the upper airway and lungs, respecting the kidneys. This represents approximately 25% of the cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In the otorhinolaryngological area, it mainly involves the paranasal sinuses and nose. Otological compromise is described in up to 38% of the cases, with ME involvement being more frequent than that of the inner ear (IE). When the IE is involved, the anterior labyrinth anterior is usually compromised and, exceptionally, the posterior. Sensorineural hearing loss (SNHL) occurs in 8% of the cases<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> and progression to cophosis is rare.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Nevertheless, in a series of 19 patients diagnosed with WG by biopsy, SNHL was observed in 12 of the 13 patients with auditory symptoms.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Theories are proposed for this disorder, such as immune complexes, granulomatous compression of the cochlea, vasculitis of the vasa vasorum<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> or nerve damage from polyneuritis.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In a recent review on otorhinolaryngological manifestations in 25 patients with WG carried out by Morales-Angulo et al.,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> not a single case of vestibular involvement was described. In less recent literature we have found only a few cases that compromise both the anterior and posterior labyrinths and none with total cochleovestibular compromise. This patient presents extraordinary characteristics of almost total cochleovestibular involvement and, based on the MRI findings, the lesion would be located at the level of the IE. Cyclophosphamide is the most effective treatment and is generally the initial therapy, associated with corticoids.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> However, due to its powerful side effects and to the fact that there was not generalized WG in our patient, azatioprina was used.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The literature describes one case of WG with bilateral moderate-severe SNHL, treated with unilateral CI, in which a mean auditory threshold of 40<span class="elsevierStyleHsp" style=""></span>db and two-syllable discrimination of 20% were obtained in free field audiometry. Our patient has achieved a level of discrimination that lets her function acceptably well in daily life. The CI would be a therapeutic possibility for rehabilitating cases of SNHL in WG.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The case presented here is very rare and atypical, not previously described in the literature, of a form of localized WG, with bilateral ear involvement. The patient displayed severe vestibular hypofunction with bilateral cophosis as the main symptoms. Knowing the atypical presentations is important to avoid delay in diagnosis and treatment, and consequently avoid possible sequelae of WG itself.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of Interests</span><p id="par0020" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical Case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of Interests" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2012-05-02" "fechaAceptado" => "2012-08-03" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Tulsidas-Mahtani B, Pérez-Garrigues H, Muñoz-Fernández N, Morera C. Afectación vestibulococlear grave en granulomatosis de Wegener localizada: una presentación atípica. Acta Otorrinolaringol Esp. 2014;65:131–132.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 569 "Ancho" => 1029 "Tamanyo" => 84676 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">3D T-2 weighted FIESTA MRI sequence of the labyrinth. MIP reconstruction of IE: damage to the basal turn of the cochlea (*) and lateral semicircular canal (#) with absence of normal hyperintensity.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 770 "Ancho" => 1028 "Tamanyo" => 214978 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histopathology. Biopsy of ME and cavum: infiltrate of chronic inflammatory cells, small foci of necrosis, fibrinoid degeneration, granulation tissue and extravasation of red blood cells (hematoxylin–eosin staining, 40×).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clinical manifestations of respiratory tract involvement in Wegener's granulomatosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "E. 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John" 3 => "M.D. Stone" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:2 [ "tituloSerie" => "UpToDate 2010" "fecha" => "2009" ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0015" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Initial immunosuppressive therapy in Wegener's granulomatosis and microscopic polyangiitis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "H. John" 1 => "M.D. Stone" 2 => "M.P.H. Andre" 3 => "A. 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Journal Information
Vol. 65. Issue 2.
Pages 131-132 (March - April 2014)
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Vol. 65. Issue 2.
Pages 131-132 (March - April 2014)
Case Study
Severe Vestibulocochlear Involvement in Wegener's Granulomatosis: A Rare Presentation
Afectación vestibulococlear grave en granulomatosis de Wegener localizada: una presentación atípica
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Bharti Tulsidas-Mahtani
, Herminio Pérez-Garrigues, Noelia Muñoz-Fernández, Constantino Morera
Corresponding author
Departamento de Otorrinolaringología, Hospital Universitario y Politécnico La Fe, Valencia, Spain
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