Chronic cough and pneumonitis secondary to cerebrospinal fluid leak: Resolution after repair

Scholfield, Daniel William; Rennie, Catherine; Saleh, Hesham A.

doi:10.1016/j.otoeng.2018.11.005

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"https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573520300090?idApp=UINPBA00004N" "url" => "/21735735/0000007100000001/v1_202002150657/S2173573520300090/v1_202002150657/en/main.assets" ] "en" => array:16 [ "idiomaDefecto" => true "cabecera" => "Case study" "titulo" => "Chronic cough and pneumonitis secondary to cerebrospinal fluid leak: Resolution after repair" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "59" "paginaFinal" => "60" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Daniel William Scholfield, Catherine Rennie, Hesham A. 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" => "*" "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" => "CT sinuses of Patient B, demonstrating a left roof of the ethmoid sinuses defect (arrow)." ] ] ] "textoCompleto" => "Case reportPatient AA 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.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 10mm diameter). Beta-2 transferrin test was positive.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>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 6mm×2mm 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">1</a> Post-operatively the patient was managed by strict bed rest with elevation of the head for 72h, a lumbar CSF drain at 5ml/h and antibiotic prophylaxis.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.Patient BA 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.<elsevierMultimedia ident="fig0010"></elsevierMultimedia>The patient proceeded to endoscopic skull base repair aided by intraoperative navigation. Intrathecal fluorescein helped identify the skull base defect, which measured 12mm 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 5ml/h.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.DiscussionOur 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.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">1</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">2</a> Fibrin sealant Tisseel (Baxter Healthcare Corporation, Illinois, USA) has been shown to reduce leak rates<a class="elsevierStyleCrossRef" href="#bib0035">3</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.The normal range of CSF pH is 7.28–7.32,<a class="elsevierStyleCrossRef" href="#bib0040">4</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.FundingThis research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Conflict of interestThe authors declare that they have no conflicts of interest." 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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." 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Acta Otorrinolaringológica Española, the official publication of the Spanish Society of Otolaryngology and Head and Neck Surgery and the Ibero-American Society of Otorhinolaryngology, is the most important journal in Spanish dedicated to Otorhinolaryngology, which reflects the continuous scientific and technical progress of the specialty. It has become an important tool to be kept up to date. The Journal publishes Original Articles (articles on basic research and clinical investigation), reviews, brief communications, case studies and images, which are subjected to a rigorous peer review process.

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