Ann Thorac Surg 2009;87:1262-1264. doi:10.1016/j.athoracsur.2008.08.058
© 2009 The Society of Thoracic Surgeons
Case Reports
Pancreaticopleural Fistula Presenting as Right-Sided Hemothorax
Arman Kilic, BS,
Chris Cook, MD,
Irfan Qureshi, MD,
Rodney J. Landreneau, MD,
Matthew J. Schuchert, MD*
Heart, Lung, and Esophageal Surgery Institute, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania
Accepted for publication August 26, 2008.
* Address correspondence to Dr Schuchert, University of Pittsburgh Medical Center, Shadyside, 5200 Centre Ave, Ste 715, Pittsburgh, PA 15232 (Email: schuchertmj{at}upmc.edu).
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Abstract
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Pancreaticopleural fistula represents a rare complication of pancreatitis. We report the case of a pancreaticopleural fistula presenting as recurrent right-sided hemothorax in a 43-year-old man. The patient was successfully treated with open surgical drainage and decortication, followed by octreotide, total parenteral nutrition, and stent placement in the pancreatic duct.
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Introduction
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Pancreaticopleural fistulas are rare complications, estimated to occur in only 0.4% of patients with pancreatitis and 4.5% of patients with pancreatic pseudocysts [1]. They are predominately found in alcoholic middle-aged men, who present with dyspnea and left-sided effusion [1, 2]. We describe a case of a pancreaticopleural fistula presenting as a right-sided hemothorax.
A 43-year-old man presented with progressive dyspnea of 2 week's duration. He had experienced a similar episode 1 month earlier when a 5-L right-sided hemothorax of unclear cause was drained with a chest tube and resolved. He denied any current fever, weight loss, nausea/vomiting, cough, hemoptysis, wheezing, or trauma. His medical history included alcohol-induced pancreatitis 7 years prior and mild reactive airway disease. He denied liver disease or a bleeding disorder. He had no significant family history. He had a 15 pack-year smoking history and denied alcohol or drug use.
The physical examination revealed an uncomfortable man who was tachycardic and tachypneic, with an oxygen saturation of 83% that improved to 98% on nasal cannula.
The results of initial laboratory data were normal except for a hemoglobin value of 6.7 g/dL, for which the patient received a transfusion. The result of a chest roentgenogram was consistent with a recurrent right-sided effusion (Fig 1).
A chest tube was placed in the right hemithorax and drained 4 L of blood. The patient was admitted for chest tube management. Results of subsequent laboratory studies revealed elevated concentrations of serum amylase and lipase. Results of liver function studies and right upper quadrant ultrasonography were normal. An abdominal computed tomography (CT) scan revealed several pancreatic pseudocysts (Fig 2).
Owing to persistent chest tube outputs and evidence of retained clot, it was decided that the patient should undergo video-assisted thoracoscopic surgical (VATS) drainage both as a diagnostic and therapeutic measure. Large clots of blood were present within the pleural space, but no active bleeding was identified intraoperatively. A sample of pleural fluid was sent for more detailed analysis and demonstrated an elevated amylase level. Results for Gram stain and cytology were negative. Analysis of pleural biopsy specimens was negative for malignancy. With continued drainage, however, the patient's amylase and lipase levels normalized and the output decreased; therefore, the chest tubes were removed serially. He was discharged on day 5.
The patient returned 4 weeks later with dyspnea secondary to recurrent right-sided hemothorax. The patient again received a transfusion and was admitted for chest tube management. Given his recent VATS procedure, the expected adhesions, and the need for more thorough exploration of the right chest, he underwent a right thoracotomy. As expected, extensive adhesiolysis was necessary. Copious amounts of bloody fluid were evacuated, and decortication was performed; however, no fistula was visualized. Fluid analysis revealed an amylase level of 102,000 U/L and lipase of 1,017,000 U/L. These extremely elevated levels established the possibility of a pancreaticopleural fistula, despite the negative intraoperative finding.
An endoscopic retrograde cholangiopancreatography (ERCP) was performed that demonstrated the pancreaticopleural fistula (Fig 3). A 5F 15-cm-long stent was placed in the pancreatic duct across the fistula, and the patient was started on octreotide therapy and total parenteral nutrition.
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Fig 3. Endoscopic retrograde cholangiopancreatography demonstrates the pancreaticopleural fistula (arrows).
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Chest tube output decreased, and the fluid amylase and lipase levels normalized. The chest tubes were removed serially, and the patient was discharged after a 2-week hospital stay. A follow-up chest roentgenogram demonstrated resolution of the hemothorax (Fig 4). A repeated ERCP 6 weeks later demonstrated closure of the fistula; however, a pancreatic leak was observed and the stent was upsized to 7F. An ERCP 2 months later demonstrated resolution of the leak.
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Comment
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A pancreaticopleural fistula presenting as hemothorax is extremely rare, having been reported once previously [3]. Our case is particularly unique in that the hemothorax was right-sided. A possible pathogenesis for this presentation is that the pancreatic enzymes entering into the chest erode the pleural lining and disrupt subpleural vessels, causing bleeding into the pleural space. Other potential causes that need to be considered in this clinical setting include trauma, spontaneous hemopneumothorax, and malignancy.
In a patient with pleural effusion and a history of pancreatitis, a pancreaticopleural fistula enters into the differential diagnosis and warrants early quantification of amylase levels in the pleural fluid. With an estimated sensitivity of 79%, ERCP is the gold standard for diagnosis, although CT is able to detect approximately 43% of cases [2]. This case report highlights the importance of early diagnosis and appropriate intervention. The thoracic operations performed might have been prevented had an ERCP been done earlier, although it could be argued that the bloody nature and large volume of pleural fluid warranted an operation to rule out an active source of bleeding or an underlying malignancy as well as to provide adequate drainage of the pleural space.
Endoscopic stent placement in the pancreatic duct is the cornerstone of treatment because it establishes a pathway of least resistance for the flow of pancreatic secretions into the duodenum, thereby excluding the fistula and facilitating healing. This is combined with therapies such as octreotide and total parenteral nutrition that are aimed at reducing pancreatic secretory activity. Chest tubes are used to manage respiratory symptoms and prevent complications but should be removed as soon as possible to prevent reduced resistance to flow through the fistula. In cases where the fistula persists, distal pancreatectomy or pancreaticojejunostomy may be performed for definitive therapy [2].
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References
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- Rockey DC, Cello JP. Pancreaticopleural fistula. Report of 7 patients and review of the literature. Medicine (Baltimore) 1990;69:332-344.[Medline]
- Oh YS, Edmundowicz SA, Jonnalagadda SS, Azar RR. Pancreaticopleural fistula: report of two cases and review of the literature Dig Dis Sci 2006;51:1-6.[Medline]
- Boudaya MS, Alifano M, Baccari S, Regnard J. Hemothorax as the clinical presentation of a pancreaticopleural fistula: report of a case Surg Today 2007;37:518-520.[Medline]
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Abstract
Introduction
