Bilothorax as a rare sign of intra-abdominal bile leak in a patient without peritonitis

Abstract

We present the case of a 60-year-old patient who developed a right-sided bilothorax following para-aortic lymph node sampling. Bilothorax is a very rare condition, with most of the reported cases resulting from direct flow of bile from the biliary system to the pleural space through a fistula or other defect in the diaphragm. In this case, there was no obvious communication found between peritoneal and pleural spaces. Our patient proceeded to an exploratory laparotomy following this finding in the absence of peritonitis. Bilothorax should be considered in patients who develop a progressive pleural effusion following upper gastrointestinal surgery, particularly if it involves manipulation of the biliary system.

Case history

A 60-year-old woman was referred to the hepatobiliary unit with a history of abdominal pain, night sweats and weight loss. Investigations in the referring unit revealed a large multicystic mass in the head of the pancreas, consistent with a possible neoplasm. As this large mass was not accompanied by jaundice, a potential diagnosis of lymphoma was considered in addition to adenocarcinoma. Following discussion at the multidisciplinary meeting, the patient was admitted for a laparotomy with a view to lymph node sampling or proceeding to a Whipple procedure.

At trial dissection, quite marked lymphadenopathy was noted in the para-aortic region, which had not been appreciated fully on preoperative staging. A sample of nodal tissue was sent for frozen section intraoperatively, the results of which indicated a lymphoma. In light of this, further lymph node harvest was performed to allow typing but no resection of the head of the pancreas was carried out. The postoperative plan was for referral to the haematology service.

The patient had a stable postoperative course until day 7 when she experienced epigastric pain and vomiting. Examination of the abdomen was unremarkable.Initial investigations included a chest radiograph, which demonstrated atelectasis and small bibasal pleural effusions. Chest physiotherapy and mobilisation were encouraged. Over the next four days, she complained of increasing dyspnoea with a modest rise in inflammatory markers despite remaining apyrexic and haemodynamically normal. Serial abdominal examinations found mild tenderness in the right upper quadrant and epigastrium with no signs of peritonism.Examination of the chest demonstrated a clinical right-sided pleural effusion.

Computed tomography was performed on the basis of this failure to progress.This demonstrated a large right-sided pleural effusion and generalised ascites (Fig 1).Given that the patient had a lymph node harvest,it was considered that the fluid in the abdomen may represent a chylous ascites.

Figure 1.

Coronal computed tomography demonstrating right-sided bilious pleural effusion (star) and ascites (arrow)

With a view to improving the patient’s dyspnoea, thoracocentesis was performed on the right hemithorax.Aspiration revealed 1 litre of bilious type fluid. Biochemical analysis of this fluid revealed a bilirubin level of 217µmol/l compared with her serum bilirubin of 39µmol/l and a diagnosis of bilothorax was made.

On the basis of this,despite the absence of peritonitis, the patient proceeded to a laparotomy. The laparotomy revealed a perforation in the Hartmann’s pouch of the gallbladder, resulting in a bilious ascites. The cause of perforation to the Hartmann’s pouch was not clear but it was speculated that it may have resulted from a thermal injury during the initial mobilisation at the trial pancreatic dissection.A cholecystectomy was performed and the abdomen washed out. Postoperatively, the patient recovered well and was transferred to the haematology department for further treatment of her lymphoma.

Discussion

Bilothorax is a very rare condition with few cases recorded in the literature in humans. Most of the reported cases result from direct flow of bile from the biliary system to the pleural space through a fistula or other defect in the diaphragm.¹ In this case, there was no obvious communication found between peritoneal and pleural spaces. This points to a more unusual cause for this patient’s bilothorax. Cases of pleural effusion in association with ascites have been reported in other conditions such as Meigs syndrome, pseudo-Meigs syndrome and hepatic hydrothorax. In all these instances, the pleural effusion resolved when the causative condition was treated definitively.²

Hypotheses to explain the effusion include spread via the lymphatics or through gaps in the muscle fibres and bundles of the diaphragm.³ In this patient, para-aortic lymph nodes had been resected, leading to a disruption in the normal lymphatic anatomy. No direct communication between the peritoneal cavity and pleura was observed during the laparotomy, and so spread between the two spaces via the lymphatics seems the most plausible explanation.

Bilious pleural effusion is a rare condition that can be a very serious cause of morbidity and mortality. It should be suspected in patients who develop a pleural effusion following upper gastrointestinal surgery, particularly if it involves manipulation of the biliary system. Symptomatic relief can be provided by draining the effusion and prescribing broad-spectrum antibiotics.However, definite management of the cause is mandatory. In our case, it is important to note that at no stage did the patient develop peritonitis, such is the insidious nature of bile in the peritoneal space. Surgeons are always suspicious that the development of a unilateral pleural effusion marks infradiaphragmatic pathology. We would recommend particular vigilance if the patient has had any procedure involving the hepatobiliary tract.

Conclusions

This case not only demonstrates the insidious nature of bile peritonitis but the unusual complication of bilothorax. The combined management of this patient between the surgeons and our colleagues in respiratory medicine enabled her to make a full recovery, proceeding to chemotherapy for lymphoma.