Abstract
INTRODUCTION
Pancreaticopleural fistula is rare. It occurs as a complication in acute and chronic pancreatitis. Here we report a case of persistent unilateral pleural effusion secondary to pancreaticopleural fistula.
PRESENTATION OF CASE
A 37 year old non alcoholic gentleman who had no history of pancreatitis and presented with breathlessness and tachypenia. X-ray chest showed massive pleural effusion on the right side. Amylase estimation of the tapping fluid was very high. ERCP showed a pancreaticopleural fistula.
DISCUSSION
Pancreaticopleural fistula is a rare entity with an incidence of 0.4-4.5%. It occurs either as a complication in pancreatitis, or after injury of the pancreatic duct. A greatly elevated pleural fluid amylase is usually the first step towards the diagnosis. ERCP and CT will identify the fistulous tract in 70%. Treatment is mainly directed towards intercostal drainage and control of the fistula.
CONCLUSION
Presentation is misleading in most of cases and needs aware clinicians with a high index of suspicion.
Keywords: Pancreaticopleural fistula, Pleural effusion, Pancreatic disease
1. Presentation of case
This 37 year old gentleman was admitted to the hospital with a 10 day history of breathlessness which was progressively getting worse. There was no history of cough or fever. He complained of some abdominal discomfort. Examination showed that he was tachypnoeic but haemodynamically stable. Abdominal examination revealed mild tenderness in the upper abdomen but no guarding. Examination of the chest revealed dullness on percussion and decreased air entry on the right side of the entire chest. Investigations showed mild elevation of the serum amylase (402 U/L). X-ray chest showed massive plueral effusion on the right side. A CT scan chest also showed a massive plural effusion but no lung lesion. U/S scan of the abdomen was unremarkable. A CT scan of the abdomen revealed dilatation of the main pancreatic duct with some calcification in the head of the pancreas. He was seen by the chest physician who tapped about 2 L of straw coloured fluid which was sent for microscopic examination. The fluid was negative for bacteria, malignant cells and red blood cells. The fluid rapidly accumulated the next day and a tap was repeated when 2.5 L of fluid was aspirated. The fluid rapidly reaccumulated the next day and an under water seal intercostal drainage tube was inserted. The fluid was sent for the usual analysis as well as for amylase estimation which showed a very high amylase (123,500 U/L). A working diagnosis of chronic pancreatitis with a pancreaticoplural fistula was made with a right sided plural efussion. Somatostatin was started and continued for 17 days along with TPN which failed to control the fistula with a continuous drainage through the intercostal tube, MRCP showed mild dilatation of the pancreatic duct suggestive of chronic pancreatitis, then patient had an ERCP which showed a fistula (Fig. 1) and had a stent inserted in the pancreatic duct on the third week since admission. The intercostal tube stopped draining on the second day after the stent insertion. He developed a loculated hydropnemothorax for which a per cutaneous drainage was attempted and then had a formal thoracotomy and decortications. This proceeded uneventfully. He was discharged on the 14th post operative day. On discharge he was afebrile, haemodynamically stable, breathing and eating well. Chest radiograph showed the lungs were fully expanded and a CT abdomen showed the stent in position.
Fig. 1.
ERCP showing fistulous track extending from the region of pancreas (junction of neck and body) superiorly into the thorax in right paraspinal location.
2. Discussion
Pancreaticopleural fistula is a rare entity with an incidence of 0.4–4.5%. It is a condition in which pancreatic secretions drain directly into the pleural cavity. It occurs either as a complication in acute and chronic pancreatitis, or after traumatic or surgical disruption of the pancreatic duct.1,2
It needs a high index of suspicion, in those with a history of acute pancreatitis and alcohol abuse, presenting with a pleural effusion, which reforms relatively rapidly after aspiration and for which there is no obvious cause. (Our case is a non alcoholic man who had no past history of pancreatitis). Majority of the cases occurred in men with chronic alcoholism aged 40–50 years. Half of the patients have no history of pancreatitis.3
Internal pancreatic fistula results from posterior disruption of the pancreatic duct into the retroperitoneal space, leading to the formation of a fistulous tract between the pancreas and the mediastinum through oesophageal hiatus.3
Diagnosis is usually made by a thoracentesis, after chest X-ray, with a greatly elevated pleural fluid amylase and lipase levels and high albumin content.4 Magnetic resonance cholangiopancreatography (MRCP) can demonstrate pancreatic pathology and the fistula. It is a non-invasive alternative to ERCP and is useful where ERCP fails to give adequate information. ERCP and CT will identify the actual fistulous tract in 70%.2
Initial treatment should be directed towards control of the pleural effusion. Intercostal tube drainage, parenteral nutrition and treatment with the somatostatin analogue octreotide achieved a 48% fistula closure rate over a 2–3 week period. Pancreatic duct stenting should be considered if the fistula fails to heal within 2–3 weeks.5
3. Conclusion
Our report highlights the rare and unusual presentation of a persistent unilateral pleural effusion in absence of a local chest aetiology. The minimal or no abdominal symptoms add difficulty to the diagnosis of the cause. Clinicians should be aware of this rare presentation with a high index of suspicion.
Conflict of interest statement
None.
Funding
None.
Ethical approval
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Author contributions
Mr Hesham El Beialy, was responsible for data collection and analysis, reviewing literature and writing up the paper.
Mr Ivo Fernandez, has laid the design and contributed to the contents of the paper.
Acknowledgement
We acknowledge the participation of Mr. Praful Vaidya2 (Consultant General Surgery), Dr. Adel Bediwy (Consultant Pulmonology), Dr. Farah Amiri2 (Consultant Radiodiagnosis) and Dr. Kameela Al-Majed2 (Resident Officer General Surgery) in the clinical management of the case.
Footnotes
International Hospital of Bahrain, P.O. Box 1084, Manama, Bahrain, www.ihb.net.
References
- 1.Schoefl R., Haefner M., Pongratz S., Pfeffel F., Stain C., Poetzi R., Gangl A. Endoscopic treatment of fistulae and abscesses in pancreatitis: three case reports. Endoscopy. 1996;28:776–779. doi: 10.1055/s-2007-1005604. [PMID 9007433] [DOI] [PubMed] [Google Scholar]
- 2.Hastier P., Rouquier P., Buckley M., Simler J.M., Dumas R., Delmont J.P. Endoscopic treatment of Wirsungo-cysto-pleural fistula. European Journal of Gastroenterology & Hepatology. 1998;10:527–529. doi: 10.1097/00042737-199806000-00017. [PMID 9855072] [DOI] [PubMed] [Google Scholar]
- 3.Dhebri A.R., Ferran N. Nonsurgical management of pancreaticopleural fistula. Journal of the Pancreas (Online) 2005;6:152–161. [PubMed] [Google Scholar]
- 4.Miller J.A., Maldjian P., Seeff J. Pancreaticopleural fistula. An unusual cause of persistent unilateral pleural effusion. Clinical Imaging. 1998;22:105–107. doi: 10.1016/s0899-7071(97)00078-8. [PMID9543587] [DOI] [PubMed] [Google Scholar]
- 5.Sudhir Kumar Jain, Baljeet Kumar Basra, Gitika Nanda, Srivathsan R., Kaza R.C.M. Rare case of internal pancreatic fistula in a young adult presenting with massive bilateral pleural effusion. BMJ Case Reports. 2009 doi: 10.1136/bcr.06.2008.0341. [pii: bcr06.2008.0341, Epub 2009 Feb 26] [DOI] [PMC free article] [PubMed] [Google Scholar]