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Saleh" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Daniel William" "apellidos" => "Scholfield" "email" => array:1 [ 0 => "danwscholfield@doctors.org.uk" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Catherine" "apellidos" => "Rennie" ] 2 => array:2 [ "nombre" => "Hesham A." "apellidos" => "Saleh" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "ENT Department, Charing Cross Hospital, Imperial Healthcare NHS Trust, London, United Kingdom" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tos crónica y neumonitis secundaria a fuga de líquido cefalorraquídeo: resolución después de la reparación" ] ] "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" => 482 "Ancho" => 900 "Tamanyo" => 32271 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">CT sinuses of Patient B, demonstrating a left roof of the ethmoid sinuses defect (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case report</span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Patient A</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 54-year-old lady was referred to our tertiary rhinology and skull base clinic with a nine-month history of right sided clear rhinorrhoea, exacerbated by leaning forward and associated with frontal headache. She had a two-year history of chronic non-productive cough and dyspnoea on exertion. There was no history of head trauma or sinonasal surgery and she had a BMI of 29. Two years previously she was hospitalised with an episode of meningitis. The source of leak could not be identified by nasendoscopy.</p><p id="par0010" class="elsevierStylePara elsevierViewall">1-mm thickness high-resolution computed tomography (HRCT) of the paranasal sinuses demonstrated an ill-defined right roof of the ethmoid sinuses <a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>, left), with loss of aeration of the right ethmoidal complex. No soft tissue mass lesions were demonstrated along the floor of the anterior cranial fossa and CT head ruled out pneumocranium. Magnetic resonance imaging (MRI) sinuses confirmed these findings, with high signal extending through from the CSF below the frontal lobes down into the right mid-ethmoid sinuses. CSF extended into the pituitary fossa with some flattening of the pituitary gland, in keeping with partially empty sella, which is associated with idiopathic intracranial hypertension. A CT thorax showed pneumonitis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>, right), with patches of ground-glass changes in both lungs and small volume hilar lymphadenopathy (up to 10<span class="elsevierStyleHsp" style=""></span>mm diameter). Beta-2 transferrin test was positive.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The patient underwent an endoscopic skull base repair aided by intraoperative navigation, after septoplasty for access. Intrathecal fluorescein was used to aid identification of the skull base defect, which measured 6<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>mm in the olfactory area. Three-layered anatomical repair was undertaken with fascia lata, septal cartilage and middle turbinate mucosa, supported by Nasopore packing (Polyganics, Gronigen, The Netherlands).<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> Post-operatively the patient was managed by strict bed rest with elevation of the head for 72<span class="elsevierStyleHsp" style=""></span>h, a lumbar CSF drain at 5<span class="elsevierStyleHsp" style=""></span>ml/h and antibiotic prophylaxis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Two weeks post-surgery the patient's cough had entirely resolved. The repair remained robust on endoscopic examination at 12-month review and was subsequently discharged from follow up.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Patient B</span><p id="par0025" class="elsevierStylePara elsevierViewall">A 55-year-old female presented with a seven-month history of left sided unilateral clear rhinorrhoea, chronic dry cough and dyspnoea on exertion. She had no history of head trauma or sinonasal surgery and had a BMI of 28. Flexible nasendoscopy was unremarkable but Beta-2 transferrin test was positive. CT sinuses demonstrated a skull base defect in the left roof of the ethmoid sinuses (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and MRI did not show any mass lesions.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The patient proceeded to endoscopic skull base repair aided by intraoperative navigation. Intrathecal fluorescein helped identify the skull base defect, which measured 12<span class="elsevierStyleHsp" style=""></span>mm in length in the olfactory area. Three-layered anatomical repair was undertaken with fascia lata from the right thigh, septal cartilage and middle turbinate mucosa. The nose was packed with Nasopore (Polyganics, Gronigen, The Netherlands) and lumbar CSF drain inserted at 5<span class="elsevierStyleHsp" style=""></span>ml/h.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The patient's cough resolved two weeks post-operatively and she had no relapse of respiratory symptoms. She was discharged from follow-up after twelve months.</p></span></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Our case series shows that CSF leak can cause pneumonitis, chronic cough and dyspnoea. Without the classical symptoms this may lead to a delay in presentation, diagnosis and subsequent repair.</p><p id="par0045" class="elsevierStylePara elsevierViewall">As in the cases described, preoperative HRCT of the sinuses is effective in localising skull base defects and intraoperative image guidance helps identification. MRI was also used due to the spontaneous nature of the leaks, as meningoencephalocele are more common amongst this demographic.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> The addition of intrathecal fluorescein aids the intraoperative localisation of CSF leak and helps to confirm an effective closure. At our institution, anatomic three-layer closure has an 89% success rate after first surgery and 100% after re-do procedures.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> Fibrin sealant Tisseel (Baxter Healthcare Corporation, Illinois, USA) has been shown to reduce leak rates<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a> and was applied between fascia lata and septal cartilage, and superficial to mucosa. The repair, as described previously by the senior author, mimics original anatomy and resulted in lasting resolution of CSF fistula in both patients. Lumbar drains were implemented in both cases due to signs of raised intracranial pressure.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The normal range of CSF pH is 7.28–7.32,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a> which is alkaline. We hypothesise that chronic exposure to CSF results in pneumonitis and chronic cough. Sealing the skull base defect thus resolves these respiratory symptoms. Further prospective and histological studies are required to further understand the histopathology behind the condition.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Funding</span><p id="par0055" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Conflict of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:5 [ 0 => array:3 [ "identificador" => "sec0005" "titulo" => "Case report" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Patient A" ] 1 => array:2 [ "identificador" => "sec0015" "titulo" => "Patient B" ] ] ] 1 => array:2 [ "identificador" => "sec0020" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0025" "titulo" => "Funding" ] 3 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflict of interest" ] 4 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-07-23" "fechaAceptado" => "2018-11-09" "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" => 522 "Ancho" => 1250 "Tamanyo" => 76890 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Left: HRCT of the paranasal sinuses of Patient A, demonstrating an ill-defined right roof of the ethmoid sinuses (arrow). Right: CT thorax of Patient A, showing ground-glass features of pneumonitis.</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" => 482 "Ancho" => 900 "Tamanyo" => 32271 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">CT sinuses of Patient B, demonstrating a left roof of the ethmoid sinuses defect (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Optimum imaging and diagnosis of cerebrospinal fluid rhinorrhoea" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "V.J. Lund" 1 => "L. Savy" 2 => "G. Lloyd" 3 => "D. Howard" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1258/0022215001904572" "Revista" => array:6 [ "tituloSerie" => "J Laryngol Otol" "fecha" => "2000" "volumen" => "114" "paginaInicial" => "988" "paginaFinal" => "992" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/11177378" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0030" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Optimising outcomes in the management of spontaneous cerebrospinal fluid rhinorrhoea" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J.S. Virk" 1 => "B. Elmiyeh" 2 => "C. Stamatoglou" 3 => "H.A. Saleh" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.4193/Rhino13.008" "Revista" => array:6 [ "tituloSerie" => "Rhinology" "fecha" => "2013" "volumen" => "51" "paginaInicial" => "268" "paginaFinal" => "274" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23943736" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0035" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Use of Tisseel fibrin sealant in neurosurgical procedures: incidence of cerebrospinal fluid leaks and cost-benefit analysis in a retrospective study" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Kassam" 1 => "M. Horowitz" 2 => "R. Carrau" 3 => "C. Snyderman" 4 => "W. Welch" 5 => "B. Hirsch" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Neurosurgery" "fecha" => "2003" "volumen" => "52" "paginaInicial" => "1102" "paginaFinal" => "1105" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/12699553" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0040" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:1 [ "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "C.A. Burris" 1 => "E.R. Ashwood" 2 => "D.E. Burns" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Libro" => array:6 [ "edicion" => "4th ed." "fecha" => "2006" "paginaInicial" => "962" "paginaFinal" => "967" "editorial" => "Elesvier Saunders" "editorialLocalizacion" => "St. Louis" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00016519/0000007100000001/v1_202001211617/S0001651919300378/v1_202001211617/en/main.assets" "Apartado" => array:4 [ "identificador" => "5841" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Caso Clínico" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00016519/0000007100000001/v1_202001211617/S0001651919300378/v1_202001211617/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001651919300378?idApp=UINPBA00004N" ]
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