BILOTHORAX: Diagnostic and Therapeutic Approaches in a Rare Case - A Clinical Review
Mohammed Quader Naseer1* and Iqra Hoor2
1mohammed Quader Naseer Medicine and Surgery, Ayaan Institute of Medical Sciences, Hyderabad, India
2Iqra Hoor Medicine and Surgery, Post Graduate (Pathology), Osmania Medical College, Hyderabad, India
*Corresponding author
Mohammed Quader Naseer, Medicine and Surgery, Ayaan Institute of Medical Sciences, Hyderabad, India.
ABSTRACT
Background: Bilothorax, or cholethorax, is a rare and serious condition where bile accumulates in the thoracic cavity, often due to complications from hepatobiliary procedures. This report examines a case managed at our institution and reviews the relevant literature to enhance understanding and management strategies.
Keywords: Bilothorax, Cholethorax, Thoracic Surgery, Bile Leakage, Case Report, Pancreatic Cancer, Pleural Effusion
Introduction
Bilothorax, also referred to as cholethorax, is an uncommon condition where bile accumulates in the thoracic cavity, requiring immediate medical attention. This report details a case of bilothorax treated at our facility and reviews related literature to improve understanding and management of this rare condition.
Case Presentation
A 62-year-old man with a history of pancreatic cancer diagnosed in early 2021 presented with obstructive jaundice. Imaging showed an enlarged pancreatic mass and increasing biliary dilatation. An attempt at endoscopic retrograde cholangiopancreatography (ERCP) for drainage was unsuccessful. An 8-French biliary drainage catheter was then inserted percutaneously, with the pigtail end in the biliary confluence.
Six weeks later, the patient arrived at the emergency department experiencing severe dyspnea, chest pain, and tachycardia. CT imaging revealed a large left hydropneumothorax compressing the lung and causing a mediastinal shift (Figure 1). The biliary drain was mispositioned, traversing the right pleural cavity and diaphragm into the liver (Figure 2), leading to bile accumulation in the thoracic cavity.
A right-sided chest tube was immediately placed, draining about 450 mL of bile-colored fluid (Figure 3) and alleviating the patient’s symptoms. Pleural fluid analysis confirmed bilothorax with a pleural bilirubin to serum bilirubin ratio greater than 1.
The patient was managed conservatively, with close monitoring, antibiotic therapy, and chest tube output surveillance. Interventional radiology replaced the biliary drainage catheter, positioning the new one in the right biliary duct under ultrasound guidance, and embolized the old tract to stop the bile leak. The patient’s respiratory condition improved, and the chest tube was removed five days post-procedure.
Despite the successful management of bilothorax, the patient’s condition worsened due to advanced metastatic pancreatic cancer, and he passed away three months later.
Discussion
Bilothorax, or bilious pleural effusion, is a rare condition often resulting from complications involving the gallbladder and biliary system. Causes include direct extension of bilomas, traumatic diaphragmatic perforations, percutaneous biliary procedures, spontaneous gallbladder perforations, and complications from surgeries such as cholecystectomy.
The formation of bilothorax typically involves small perforations in the diaphragm, allowing bile to flow from the peritoneal cavity into the pleural space. In percutaneous biliary interventions, the catheter may create an iatrogenic fistula. Increased pressure in the obstructed biliary system can also force bile into the pleural cavity, facilitated by negative inspiratory pressure.
Symptoms include dyspnea, cough, pleuritic chest pain, and signs of sepsis. Without treatment, respiratory deterioration can occur, potentially leading to acute respiratory distress syndrome (ARDS). Empyema, often involving gastrointestinal pathogens, is a common complication.
Diagnosis is usually straightforward but requires suspicion, particularly after hepatobiliary procedures. Chest radiographs showing pleural effusion should prompt consideration of bilothorax. Characteristic bile-colored fluid and a pleural bilirubin to serum bilirubin ratio greater than 1 confirm the diagnosis. CT scans may show fistulas between the abdominal and chest cavities, and ERCP can demonstrate bile leakage.
Management typically involves conservative measures such as chest tube drainage, antibiotic therapy, and controlling the biliary source. Invasive surgical interventions may be necessary for persistent leaks or non-responsive empyema. Minimally invasive techniques like percutaneous drainage and ERCP to relieve biliary obstruction, combined with pleural drainage, have been effective. (Table.1)
Preventative measures include intraoperative checks for bile leaks and the use of intra-abdominal drains. Early broad-spectrum antibiotics can prevent infections. Close monitoring, adequate drainage, and control of the biliary source are crucial. Interventional radiology can be instrumental in stopping bile leaks, as seen in this case.
The prognosis of bilothorax depends on the underlying condition. In cases of advanced malignancies, like the metastatic pancreatic cancer in this report, prognosis remains poor despite successful management of bilothorax. Multidisciplinary care involving thoracic surgeons, interventional radiologists, oncologists, and palliative care specialists is essential for optimal patient outcomes.
Conclusion
Bilothorax, an uncommon exudative pleural effusion, requires high suspicion for diagnosis. Common causes include iatrogenic hepatobiliary procedures, trauma, and infections. Management involves draining the pleural effusion, administering antibiotics, and addressing biliary obstructions. Early recognition and appropriate treatment are crucial to reduce morbidity and mortality associated with bilothorax.
Declarations: Ethical Approval
Human Subjects: Consent was obtained or waived by all participants in this study.
Consent to publish -also taken
Authors’ Contributions: Mohammed Quader Naseer and Mohammed Afshar Alam wrote the main manuscript text.
Iqra Hoor collected figures and wrote the cover letter-All authors reviewed the manuscript.
Funding: none recieved
Availability of Data and Materials: can be asses via mail at mdquader69@gmail.com or databases search including pubmed, scopus, web of science.
Conflicts of Interest: In compliance with the ICMJE uniform disclosure form, all authors declare the
following: Payment/Services Info
All authors have declared that no financial support was received from any organization for the submitted work.
Financial Relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work.
Other Relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work.
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