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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 9  |  Issue : 2  |  Page : 63-65

Pancreaticopleural fistula causing recurrent pleural effusion in acute pancreatitis


Department of PICU, SAACHI Children Hospital, Surat, Gujarat, India

Date of Submission24-Nov-2021
Date of Decision30-Jan-2022
Date of Acceptance07-Feb-2022
Date of Web Publication30-Mar-2022

Correspondence Address:
Dr. Vipul Chechani
B-203, Surya Palace, Citylight, Surat - 395 007, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpcc.jpcc_95_21

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  Abstract 


Digestive property intrinsic to pancreatic secretions can cut through the fascial planes and may form a pancreaticopleural fistula which is an extremely rare critical complication of pancreatitis in children. It links the pancreas with the pleural cavity through the diaphragmatic hiatus which can feed the pleural space with amylase-rich secretions and can cause recurrent pleural effusion. We report a rare case of gross left-sided pleural effusion (hemothorax) in a patient due to pancreatic etiology. The clinical presentation was deceptive since the patient presented with predominantly respiratory complaints. Many patients go through extensive pulmonary evaluation before the pancreas is recognized as the site of basic pathology. Pleural fluid amylase levels and visualizing the fistula tract from the pancreatic duct to the pleural space by radiological imaging provide the confirmation of this complication. High index of suspicion of pancreatic etiology in recurrent massive hemorrhagic pleural effusion may lead to prompt diagnosis.

Keywords: Pancreaticopleural fistula case report, pancreatitis, recurrent pleural effusion


How to cite this article:
Chechani V, Tadvi H, Bhimani U. Pancreaticopleural fistula causing recurrent pleural effusion in acute pancreatitis. J Pediatr Crit Care 2022;9:63-5

How to cite this URL:
Chechani V, Tadvi H, Bhimani U. Pancreaticopleural fistula causing recurrent pleural effusion in acute pancreatitis. J Pediatr Crit Care [serial online] 2022 [cited 2023 Feb 8];9:63-5. Available from: http://www.jpcc.org.in/text.asp?2022/9/2/63/341450




  Introduction Top


Pancreaticopleural fistula (PPF) is a very rare and critical complication of pancreatitis consequent to disruption of the pancreatic duct. The fistula track ascends into the pleural space and gives rise to large volumes of pleural fluid. The presentation is often confusing because of the lack of clues suggestive of pancreatic etiology and the predominance of pulmonary signs and symptoms. The pleural effusion is particularly rapidly accumulating, recurrent, and exudative. PPF is seen in 0.4% of patients presenting with pancreatitis in adults.[1] Most of the literature on PPF is related to chronic pancreatitis (CP) in adults and pediatric cases secondary to acute pancreatitis have not been reported.


  Case Report Top


A 3-year-old female child was brought to our emergency unit with respiratory distress and irritability for 3 days. History of hospitalization 3 weeks back for 14 days where the child had left-sided pleural effusion requiring ICD placement which was removed before discharge. At that time, the child was given intravenous antibiotics and AKT was also started. However, after 3 days of discharge, the child again had similar complaints. She had no past history of abdominal trauma, drug intake, recent infection, previous surgery, biliary, and pancreatic diseases. On admission, the child had tachypnea, tachycardia, grunting, intercostal retractions, and on auscultation decreased air entry on the left side. The chest X-ray showed gross pleural effusion on the left side with mediastinal shift to the opposite side. The left side intercostal tube was inserted from where hemorrhagic pleural fluid collected and sent for routine and microscopic examination. Computed tomography (CT) chest was performed to identify the etiology of recurrent pleural effusion where radiologist incidentally found pancreaticopleural fistula in the CT images of the upper abdomen. On CT abdomen, the pancreas was diffusely bulky and showed homogenous postcontrast enhancement. Pleural fluid amylase and lipase were 2000 and 26,000 U/L, respectively, compared to serum amylase and lipase of 1620 and 2489 U/L, respectively. The child was diagnosed having pancreaticopleural fistula as the etiology for recurring pleural effusion. For pancreatitis, the child was managed conservatively with bowel rest followed by clear fluids and then soft diet. To evaluate the biliary tract in detail, magnetic resonance cholangiopancreatography (MRCP) was done which showed walled off intra and peripancreatic collection in mid-distal body of the pancreas with disruption of main pancreatic duct. A thick fistulous tract was seen arising from the superior aspect of the collection up to gastroesophageal junction from where through the esophageal hiatus it went into the pleural space. As there was massive pleural effusion with pleural thickening and internal septations, a video-assisted thoracoscopic surgery was performed. The child showed gradual improvement over the next 7 days on conservative management. The child was given nutritional rehabilitation with protein-rich diet and discharged after 13 days of hospitalization.


  Discussion Top


PPF is a rare complication of pancreatitis. It can be caused by acute or CP, pancreatic trauma, or iatrogenic rupture of the pancreatic duct. CP related to alcohol abuse (67%) is the most common causative factor in adults.[2] In children, there is insufficient data on the incidence and causes of PPF, such as acute pancreatitis (blunt trauma and idiopathic) and CP (ductal anomalies, PRSS1 mutation, and idiopathic).

Considering that in India, tuberculosis is the most common cause of exudative pleural effusion; also based on history, clinical examination, and investigation reports, a diagnosis of tubercular pleural effusion is common initially. The average time to diagnose PPF is 5 weeks based on previous study,[1] which in our case was 3 weeks. The primary reason for delayed diagnosis is that PPF is a rare condition, and the major manifestations are pulmonary symptoms caused by repeated pleural effusion, and abdominal symptoms are uncommon.

PPF may present initially with a fast breathing, dyspnea, chest tightness, with or without a complaint of abdominal pain, or steatorrhea. PPF-related pleural effusion is usually refractory to drainage and has a tendency to accumulate rapidly. Small and self-limited leftsided effusion without elevated pancreatic enzymes is commonly seen due to increased vascular permeability secondary to diaphragmatic inflammation in acute pancreatitis. Massive recurrent exudative effusion with raised pancreatic enzymes occurs due to the development of PPF. For imaging, MRCP is the technique of choice because it is less invasive and more sensitive than CT and endoscopic retrograde cholangiopancreatography.[3]

As defined by the INternational Study Group of Pediatric Pancreatitis: in Search for a CuRE (INSPPIRE) consortium, CP requires imaging findings characteristic of and consistent with CP (specifically, radiographically evident calcifications, and pancreatic duct irregularities, such as strictures and dilations).[4] In our case, there were no imaging findings characteristic of CP and the child never had any symptoms or signs suggestive of pancreatitis in the past so it was labeled acute pancreatitis. Although PPF is more commonly seen with CP in pediatric patients.

The treatment of PPF includes conservative treatment, endoscopic intervention, or surgical management which depends primarily on the ductal anatomy. A normal or mildly dilated pancreatic duct can be managed with conservative treatment, including ICD placement, trypsin inhibitor, nasojejunal tube feeding, and total parenteral nutrition. In 30%–60% of cases, medical treatment is successful.[3],[5] In case of incomplete ductal disruption in the head or body of the pancreas and distal stricture, an endoscopic approach can be done initially using a stent, sphincterotomy, or balloon dilatation, which can decrease the pressure of the pancreatic duct. If endoscopic treatment is not feasible due to complete ductal disruption, ductal obstruction proximal to fistula, leak in the tail region, or failure of endoscopic management, surgical intervention is done, such as partial pancreatectomy, longitudinal pancreaticojejunostomy, or internal drainage of pseudocysts. PPF in children is a rare condition and so there are no relevant studies to decide which therapeutic method is the best. Surgical management is the definitive line of management for PPF. However, it is resorted to only after the failure of medical or endoscopic treatment.

Recurrent hemorrhagic pleural effusions of unknown etiology may be caused by a pancreaticopleural fistula in acute pancreatitis. It can be confirmed by estimating pleural fluid amylase level. Lack of awareness of this rare complication can result in a delay in the diagnosis and morbidity.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient's legal guardian has given the consent for images and other clinical information to be reported in the journal. The patient's legal guardian understands that name and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Tay CM, Change SK. Diagnosis and management of pancreaticopleural fistula. Singapore Med J 2013;54:190-4.  Back to cited text no. 1
    
2.
Wypych K, Serafin Z, Gałązka P, Strześniewski P, Matuszczak W, Nierzwicka K, et al. Pancreaticopleural fistulas of different origin: Report of two cases and a review of literature. Pol J Radiol 2011;76:56-60.  Back to cited text no. 2
    
3.
Ali T, Srinivasan N, Le V, Chimpiri AR, Tierney WM. Pancreaticopleural fistula. Pancreas 2009;38:e26-31.  Back to cited text no. 3
    
4.
Morinville VD, Husain SZ, Bai H, Barth B, Alhosh R, Durie PR, et al. Definitions of pediatric pancreatitis and survey of present clinical practices. J Pediatr Gastroenterol Nutr 2012;55:261-5.  Back to cited text no. 4
    
5.
Rockey DC, Cello JP. Pancreaticopleural fistula. Report of 7 patients and review of the literature. Medicine (Baltimore) 1990;69:332-44.  Back to cited text no. 5
    




 

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