Scientific Letter
Spontaneous Bilateral Chylothorax Caused by a Sneeze: An Unusual Entity with Good Prognosis
Quilotórax espontáneo bilateral desencadenado por estornudo: una entidad peculiar de buen pronóstico
Javier García-Tirado
, Hugo S. Landa-Oviedo, Iliana Suazo-Guevara
Corresponding author
Servicio de Cirugía Torácica, Hospital Universitario Miguel Servet, Zaragoza, Spain
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Landa-Oviedo, Iliana Suazo-Guevara" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Javier" "apellidos" => "García-Tirado" "email" => array:2 [ 0 => "fco854@separ.es" 1 => "jgarciat@salud.aragon.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Hugo S." "apellidos" => "Landa-Oviedo" ] 2 => array:2 [ "nombre" => "Iliana" "apellidos" => "Suazo-Guevara" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Cirugía Torácica, Hospital Universitario Miguel Servet, Zaragoza, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Quilotórax espontáneo bilateral desencadenado por estornudo: una entidad peculiar de buen pronóstico" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1481 "Ancho" => 1500 "Tamanyo" => 248332 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Axial computed tomography slices showing left laterocervical-supraclavicular collection, with diffuse extension throughout all mediastinal compartments (circled) and bilateral pleural effusion. (A) Posteroanterior radiograph of chest after removal of left pleural drainage tube, with the right pleural drainage tube still in place. (B) Posteroanterior radiograph of chest during follow-up, 3 months after the episode, showing complete resolution of bilateral pleural effusion.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Chylothorax is defined as the presence of triglycerides in pleural fluid (>110<span class="elsevierStyleHsp" style=""></span>mg/dl) with a pleural fluid/serum ratio >1, positive Sudan III staining or detection of chylomicrons. It is the most common cause of pleural effusion in fetuses and neonates, but in adults it accounts for only 3% of cases of this entity. It is mainly caused by cancer (lymphoma in 75% of cases), followed by iatrogenesis and injury. Other rare causes include lymphangiomyomatosis, sarcoidosis, tuberculosis, congenital and idiopathic lymphatic malformations.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 60-year-old patient with a history of arterial hypertension, dyslipidemia, and hypothyroidism receiving medical treatment. After a violent sneeze, she developed left supraclavicular swelling and dizziness, which prompted her to visit the emergency room. The chest radiograph revealed bilateral basal pleural effusion; cervical-chest computed tomography (CT) showed left laterocervical-supraclavicular collection extending to all mediastinal compartments, with bilateral pleural effusion (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), interpreted by the radiologist as hematoma following a violent sneeze. A wait-and-see approach was adopted, and after bilateral progression of the effusion was confirmed on X-ray, endopleural drainage tubes were placed in both sides. Fluid with a chylous appearance was obtained from both hemithoraces, and analysis of samples from both sides confirmed sterile exudate with a high triglyceride concentration (2241<span class="elsevierStyleHsp" style=""></span>mg/dl in the right hemithorax and 2160<span class="elsevierStyleHsp" style=""></span>mg/dl in the left). After diagnostic confirmation of bilateral chylothorax, dietary treatment was initiated, with restricted fat intake. Fluid collected from both drainage tubes reduced progressively, becoming serous in appearance after the introduction of dietary restrictions, confirming the biochemical resolution of the chylothorax. In view of the patient's favorable progress, no other therapeutic measures were introduced, and the pleural drainage tubes were removed from the left side after 48<span class="elsevierStyleHsp" style=""></span>h (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A) and from the right side after 5 days. After removal of the drainage tubes, radiological monitoring confirmed bilateral resolution of the effusion. Resolution of clinical and radiological signs and symptoms were confirmed in outpatient follow-up (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Minor injury triggering chylothorax has been described in the literature: forced stretching of the upper limbs, or maneuvers that increase intrathoracic pressure, including vomiting, hiccoughs after ingestion of high-fat foods, cough, childbirth or, very rarely, sneezing – as in our case. We found no other case of chylothorax caused by sneezing in either PubMed or Embase.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> Cases in which no cause of disruption of the thoracic duct can be determined on examination are considered spontaneous; however, concomitant presence of a fragile duct is very likely to be a predisposing factor.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Very few cases have been reported in the literature; a review of similar cases reveals a rather well-defined pattern, consisting of the appearance of left supraclavicular swelling along with bilateral chylothorax after mild exertion in a woman in her fifth decade of life.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> In our patient, the temporal relationship between the sneeze and the appearance of lymphatic effusion in the absence of any other specific cause led us to establish a diagnosis of spontaneous chylothorax. Performing a CT is important for the correct characterization of the clinical picture and the grade of involvement, and for detecting any underlying issue (thoracic duct disease, lymphadenopathy, masses, lung disease, etc.).<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4,5</span></a> Unlike cases caused by major traumatism, rupture of the thoracic tract is minimal in spontaneous cases and lymphography is generally inconclusive; it is unlikely that further studies other than CT and analysis of the pleural fluid, such as we performed in our case, will be required.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In general, the best treatment for chylothorax is to correct the underlying cause when it is identified.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Therapeutic management may include conservative measures (dietary restriction of fats, evacuating thoracocentesis, parenteral nutrition, treatment with octreotide), minimally invasive measures (pleural drainage, pleurodesis, interventional radiological techniques), and surgical intervention (thoracic duct ligation with video-assisted thoracoscopy or thoracotomy).<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> When the amount of fluid collected is sparse, as in spontaneous cases, chylothorax can be appropriately controlled with dietary treatment and evacuation of pleural effusion, if demanded by the volume, with evacuating thoracocentesis or pleural drainage,<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> as in our case. It is important for physicians to understand spontaneous chylothorax, as prognosis with conservative treatment is good, in contrast to chylothorax induced by other causes.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: García-Tirado J, Landa-Oviedo HS, Suazo-Guevara I. Quilotórax espontáneo bilateral desencadenado por estornudo: una entidad peculiar de buen pronóstico. Arch Bronconeumol. 2017;53:32–33.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1481 "Ancho" => 1500 "Tamanyo" => 248332 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Axial computed tomography slices showing left laterocervical-supraclavicular collection, with diffuse extension throughout all mediastinal compartments (circled) and bilateral pleural effusion. (A) Posteroanterior radiograph of chest after removal of left pleural drainage tube, with the right pleural drainage tube still in place. (B) Posteroanterior radiograph of chest during follow-up, 3 months after the episode, showing complete resolution of bilateral pleural effusion.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Traumatic chylothorax: a case report and review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "W. Sendama" 1 => "M. 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Sansano" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:2 [ "tituloSerie" => "Radiologia" "fecha" => "2016" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Quilotórax" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "N. Mañes Bonet" 1 => "H. Hernández Rodríguez" 2 => "R. Gil Lorenzo" 3 => "G. 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| 2021 July | 32 | 25 | 57 |
| 2021 June | 60 | 26 | 86 |
| 2021 May | 46 | 36 | 82 |
| 2021 April | 88 | 92 | 180 |
| 2021 March | 80 | 21 | 101 |
| 2021 February | 31 | 23 | 54 |
| 2021 January | 26 | 19 | 45 |
| 2020 December | 31 | 12 | 43 |
| 2020 November | 19 | 19 | 38 |
| 2020 October | 25 | 17 | 42 |
| 2020 September | 28 | 13 | 41 |
| 2020 August | 31 | 12 | 43 |
| 2020 July | 40 | 29 | 69 |
| 2020 June | 28 | 7 | 35 |
| 2020 May | 37 | 9 | 46 |
| 2020 April | 36 | 21 | 57 |
| 2020 March | 31 | 17 | 48 |
| 2020 February | 38 | 23 | 61 |
| 2020 January | 38 | 19 | 57 |
| 2019 December | 54 | 17 | 71 |
| 2019 November | 41 | 15 | 56 |
| 2019 October | 20 | 10 | 30 |
| 2019 September | 26 | 12 | 38 |
| 2019 August | 24 | 17 | 41 |
| 2019 July | 47 | 15 | 62 |
| 2019 June | 28 | 18 | 46 |
| 2019 May | 46 | 22 | 68 |
| 2019 April | 75 | 21 | 96 |
| 2019 March | 35 | 34 | 69 |
| 2019 February | 28 | 12 | 40 |
| 2019 January | 18 | 14 | 32 |
| 2018 December | 21 | 9 | 30 |
| 2018 November | 42 | 20 | 62 |
| 2018 October | 43 | 19 | 62 |
| 2018 September | 23 | 10 | 33 |
| 2018 May | 13 | 1 | 14 |
| 2018 April | 30 | 3 | 33 |
| 2018 March | 26 | 1 | 27 |
| 2018 February | 31 | 9 | 40 |
| 2018 January | 71 | 7 | 78 |
| 2017 December | 68 | 8 | 76 |
| 2017 November | 48 | 6 | 54 |
| 2017 October | 20 | 11 | 31 |
| 2017 September | 29 | 9 | 38 |
| 2017 August | 20 | 8 | 28 |
| 2017 July | 31 | 8 | 39 |
| 2017 June | 24 | 13 | 37 |
| 2017 May | 31 | 5 | 36 |
| 2017 April | 27 | 31 | 58 |
| 2017 January | 2 | 0 | 2 |
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