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Case Report
A Rare Presentation of Bilothorax
Andrés Briones-Gómez, María Sánchez-Samblancat, Amina Bekki
Corresponding author
amina.bekki.gva@gmail.com

Corresponding author.
Unidad de Endoscopia Respiratoria y Neumología Intervencionista, Servicio de Neumología, Hospital Universitario y Politécnico La Fe, Valencia, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0050" class="elsevierStylePara elsevierViewall">Bilothorax &#40;also called cholethorax or thoracobilia&#41; is defined as the presence of bile in the pleural fluid &#40;PF&#41; and is a rare cause of exudative pleural effusion&#46; Most of the cases described in the literature are associated with hepatobiliary surgery&#44; biliary tract intervention&#44; or biliary infections with pleurobiliary fistulas&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">1&#44;2</span></a> In almost all cases&#44; involvement was exclusively in the right pleural cavity&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 58-year-old woman with a history of perihilar cholangiocarcinoma &#40;also known as Klatskin tumor&#41; who had undergone tumor resection surgery 10 months previously followed by chemotherapy&#46; She was being monitored by Oncology and was&#44; in principle&#44; disease-free&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">She attended the emergency department for progressive dyspnea&#44; and a thoracoabdominal computed tomography &#40;CT&#41; scan revealed left pleural effusion&#46; Post-surgical complications and bile duct fistula were ruled out&#46; Chest ultrasound showed a moderate collection of pleural fluid&#44; anechoic in appearance&#44; containing fibrin tracts&#46; Diagnostic thoracentesis was performed in the left hemithorax&#44; and the fluid obtained was remarkable for its dark greenish and viscous macroscopic appearance&#44; suggesting bilothorax &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The PF study is described in <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#46; The study was completed with diagnostic medical thoracoscopy that revealed a thickened&#44; irregular and erythematous parietal pleura with hyperpigmented yellowish lesions&#46; Several pleural biopsies were obtained&#46; Pathology confirmed the suspicion of a metastatic process consistent with a relapse of perihilar cholangiocarcinoma&#46; Finally&#44; a tunneled chest tube was placed for better symptomatic management of the effusion&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Bilothorax is a rare cause of pleural exudate&#44; resulting primarily from biliary tract lesions or diseases that form pleurobiliary fistulas&#46; These include malignant or benign biliary obstruction&#44; disruption of pleural or fissure integrity following hepatobiliary procedures &#40;surgery or percutaneous drainage&#41;&#44; and hepatic or subphrenic abscesses&#46; The presence of bile in the pleural space can cause a significant inflammatory reaction and PF superinfection is common&#46; Involvement is typically right-sided due to anatomical proximity&#44; but bile can also pass naturally through the esophageal and aortic hiatuses&#44; leading to involvement of the left pleural cavity&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The most specific diagnostic criterion is a PF&#47;serum bilirubin ratio greater than 1&#46; Some authors propose glycolic acid in PF as an additional criterion&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Our case is significant for left pleural unilateral involvement in the absence of recent hepatobiliary surgery or documented pleurobiliary fistula&#46; An exhaustive review of the literature retrieved no cases of Klatskin tumor with histologically confirmed contralateral pleural metastasis manifesting as bilothorax&#44; hence the interest of the case we describe&#46; The treatment is the same as that of the cause&#58; chemotherapy&#44; in the case of our patient&#44; or closure of fistulas if the cause is iatrogenic&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors state that they have no conflict of interests&#46;</p></span></span>"
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