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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 4  |  Page : 206-208

Right-sided massive hemorrhagic pleural effusion with pancreaticopleural fistula secondary to pancreatitis: A rare cause of pediatric respiratory distress


Department of Pediatrics, Sarji Hospital, Shimoga, Karnataka, India

Date of Submission26-Mar-2020
Date of Decision17-May-2020
Date of Acceptance25-May-2020
Date of Web Publication13-Jul-2020

Correspondence Address:
Dr. H S Vinayaka
Sarji Hospital, RMR Road, Park Extension, Shimoga - 577 201, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JPCC.JPCC_42_20

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  Abstract 


Hemorrhagic pleural effusion secondary to pancreaticopleural fistula (PPF) due to acute-on chronic pancreatitis is a very rare cause of pediatric respiratory distress. Hemorrhagic pleural effusion secondary to PPF will be usually left sided and accounts for 1% of all cases; very rarely, it could be right sided resulting in difficulty in establishing the diagnosis if chest symptoms are disproportionately more than the abdominal symptoms. We report a 9-year-old male child who presented to us with severe respiratory distress, with insignificant history later found to have right-sided massive hemorrhagic pleural effusion with PPF secondary to acute-on chronic pancreatitis.

Keywords: Hemorrhagic pleural effusion, pancreaticopleural fistula, pancreatitis, respiratory distress


How to cite this article:
Vinayaka H S, Rudrappa DS, Sathish H S. Right-sided massive hemorrhagic pleural effusion with pancreaticopleural fistula secondary to pancreatitis: A rare cause of pediatric respiratory distress. J Pediatr Crit Care 2020;7:206-8

How to cite this URL:
Vinayaka H S, Rudrappa DS, Sathish H S. Right-sided massive hemorrhagic pleural effusion with pancreaticopleural fistula secondary to pancreatitis: A rare cause of pediatric respiratory distress. J Pediatr Crit Care [serial online] 2020 [cited 2020 Aug 9];7:206-8. Available from: http://www.jpcc.org.in/text.asp?2020/7/4/206/289521




  Introduction Top


Hemorrhagic pleural effusion due to pancreaticopleural fistula (PPF) is extremely rare entity accounting for <1% of cases,[1] and it has been recognized as a clinical entity since the late 1960.[2] Still, the diagnosis of hemorrhagic pleural effusion is often missed. This rare entity has been seen in patients with acute and chronic pancreatitis or may follow after surgical disruption of the pancreatic duct. It usually presents with recurrent large hemorrhagic pleural effusion on either pleural space, but left side accounts for 76% of cases.[3],[4],[5],[6],[7] Most cases of hemorrhagic pleural effusion secondary to pancreatitis were between the age of 20 and 55 years, and most of the patients are alcoholics. We report a 9-year-old male child with right-sided massive hemorrhagic pleural effusion secondary to PPF.


  Case Report Top


A 9-year-old child came to us with a history of right-sided chest pain for 1 month, progressive dyspnea for 2 weeks, and hurried breathing for 1 day. His history was insignificant. At presentation, the child had severe respiratory distress with decreased air entry on the right side and mediastinal shift toward the left side. Chest X-ray done showed right-sided complete haziness with the shift of cardia toward the left side [Figure 1]. Point-of-care ultrasound done confirmed the presence of massive right-sided effusion with internal echoes. Emergency intercostal drainage (ICD) tube was inserted, which revealed dark red bloody effusion, and drained around 2 l of hemorrhagic effusion over a period of 24 h. After ICD insertion, child respiratory distress came down.
Figure 1: Chest X-ray showing right-sided complete haziness with mediastinal shift toward the left side

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Investigations done revealed hemoglobin 10.2 g/dl, hematocrit 32%, total leukocyte count 12,000 cells/cumm, platelet count 5.8 lakhs/cumm, negative C-reactive protein, normal liver function test, and normal prothrombin time and activated partial thromboplasti time (APTT). Pleural fluid analysis done showed hematocrit of 15%, total leukocyte count of 900 cells/cumm, sugar of 47.3 mg/dl, and protein of 3.32 g/dl. Computed tomography (CT) of the thorax with abdomen was done as a part of evaluation of hemorrhagic pleural effusion. The CT confirmed the presence of chronic pancreatitis. Pleural fluid amylase done was grossly elevated 3760 μ/l (normal value 30–110 μ/l). Serum amylase was also elevated 760 μ/l (normal range 30–85 μ/l). Later, the child was subjected to magnetic resonance cholangiopancreatography (MRCP) [Figure 2], which showed features of chronic calcified pancreatitis with intra and peripancreatitic psuedocyst with right-sided PPF.
Figure 2: Magnetic resonance cholangiopancreatography showing pancreaticopleural fistula with chronic pancreatitis and peripancreatic pseudocyst formation

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After confirming the diagnosis of hemorrhagic pancreatic pleural effusion, the child was started on octreotide infusion. ICD output started reducing after octreotide infusion. Later, the child was subjected to endoscopic retrograde cholangiopancreatography (ERCP), with pancreatic duct stent placement. ICD was removed once the ICD drainage has come down. Later, the child was discharged with pancreatic supplements.


  Discussion Top


Hemorrhagic pleural effusion is often missed many times; the common causes include trauma, intrathoracic neoplasms, bleeding diathesis, rupture due to pulmonary embolism, iatrogenic interventions, and tuberculosis.[8] Pancreatic pleural effusion often occurs as a complication of acute pancreatitis, pancreatic abscess, pseudocyst, and chronic pancreatitis.[9] There are two types of pleural effusion complicated by pancreatic pathologies. The first one is usually small left-sided reactive pleural effusion with normal amylase activity (below 100) and low protein concentration (<3 g/dl); this type of effusion is usually associated with acute pancreatitis and resolves during recovery. The second one is usually large, single-sided, recurrent and contains a high level of amylase above 1000 μ/L and protein above 3 g/dl, this type of effusion is usually related to the presence of PPF in the course of chronic pancreatitis.[10],[11] The postulated pathogenic mechanism of hemorrhagic pleural effusion includes transdiaphragmatic transfer of pancreatic fluid through lymphatics, diaphragmatic perforation of pseudocyst, and mediastinal extension.[8] Few studies demonstrated that a fistula connecting a pancreatic psuedocyst with the pleural cavity was the pathogenic mechanisms of pleural effusion.[12],[13] In our case, it was right-sided massive hemorrhagic pleural effusion with elevated amylase (3760 μ/l) in the pleural fluid and elevated protein (3.32 g/dl). MRCP confirms the presence of PPF.

If the chest symptoms predominate with no previous history of pancreatic disease, diagnosis often becomes difficult and the many patients go through extensive pulmonary evaluation before the identification of pancreatic pathology. As the underlying pancreatic disease may be asymptomatic, pleural fluid amylase gives a valuable clue in case of unexplained massive hemorrhagic pleural effusion. Pleural fluid amylase will be markedly elevated (>1000 μ/l), even when the serum amylase is mildly elevated. In our case also, he presented predominantly with the chest symptoms, with no clue regarding the presence of pancreatitis. On evaluating the cause of hemorrhagic pleural effusion, CT thorax with abdomen was done. This showed the presence of features of chronic pancreatitis. Later, pleural fluid amylase was done, which showed marked elevation (3760 μ/l).

Investigation of choice for the diagnosis of PPF is MRCP because of its noninvasive nature and its ability to visualize beyond strictures. Moreover, it can also depicts parenchymal atrophy, ductal anatomy and small intrapancreatic or extrapancreatic pseudocysts, peripancreatic collections or PPF.[14]

The available treatment modalities are (1) conservative/medical management, (2) ERCP plus/minus endoscopic pancreatic stent placement, and (3) surgery.[3],[4] Treatment with drainage of pleural effusion by ICD tube, followed by conservative treatment to decrease the exocrine pancreatic secretions (nasogastric suction, total parental nutrition, and octreotide infusion), is usually effective in massive pancreatic pleural effusion.[10] If the chest tube drainage fails, percutaneous drainage of abdominal pseudocyst or gastrocystostomy can be considered for the pseudocyst. If the patient has PPF in the head or body, ERCP can be successfully treated by placing stent in the pancreatic duct.[7] Surgery is considered only if medical or endoscopic therapy fails.


  Conclusion Top


Pancreatitis should be taken into consideration if there is any evidence of hemorrhagic pleural effusion. Early pleural fluid amylase testing will avoid delayed diagnosis. MRCP is the investigation of choice to diagnose PPF. Treatment with drainage of the effusion by chest tube, inhibition of pancreatic secretions by octreotide infusion, and ERCP plus stenting the pancreatic duct is very effective in massive pancreatic pleural effusion.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Burgess NA, Moore HE, Williams JO, Lewis MH. A review of pancreatico-pleural fistula in pancreatitis and its management. HPB Surg 1992;5:79-86.  Back to cited text no. 1
    
2.
Anderson WJ, Skinner DB, Zuidema GD, Cameron JL. Chronic pancreatic pleural effusions. Surg Gynecol Obstet 1973;137:827-30.  Back to cited text no. 2
    
3.
King JC, Reber HA, Shiraga S, Hines OJ. Pancreatic-pleural fistula is best managed by early operative intervention. Surgery 2010;147:154-9.  Back to cited text no. 3
    
4.
Sut M, Gray R, Ramachandran M, Diamond T. Pancreaticopleural fistula: A rare complication of ERCP-induced pancreatitis. Ulster Med J 2009;78:185-6.  Back to cited text no. 4
    
5.
Dhebri AR, Ferran N. Nonsurgical management of pancreaticopleural fistula. JOP 2005;6:152-61.  Back to cited text no. 5
    
6.
Vyas S, Gogoi D, Sinha SK, Singh P, Yadav TD, Khandelwal N. Pancreaticopleural fistula: An unusual complication of pancreatitis diagnosed with magnetic resonance cholangiopancreatography. JOP 2009;10:671-3.  Back to cited text no. 6
    
7.
Safadi BY, Marks JM. Pancreatic-pleural fistula: The role of ERCP in diagnosis and treatment. Gastrointest Endosc 2000;51:213-5.  Back to cited text no. 7
    
8.
Raghu MB, Balasubramanian S, Balasubramanyam G. Hemorrhagic pleural effusion—Sole manifestation of pancreatitis. Indian J Pediatr 1992;59:767-9.  Back to cited text no. 8
    
9.
Sumalata C, Gopichand N. An unusual presentation of pancreatic pleural effusion. Sch J Med Case Rep 2014;2:612-4.  Back to cited text no. 9
    
10.
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. 10
    
11.
Sontakke A, Tayade BO. Case series of pancreatic pleural effusion with pancreatico-pleural fistula. JIACM 2014;15:245-8.  Back to cited text no. 11
    
12.
Imai Y, Taniguchi M, Tagawa K, Toda N, Tanzawa Y, Okamoto M, et al. A case of chronic pancreatitis complicated by massive pericardial and right pleural effusion. Gastroenterol Jpn 1993;28:734-8.  Back to cited text no. 12
    
13.
Dewan NA, Kinney WW, O'Donohue WJ Jr. Chronic massive pancreatic pleural effusion. Chest 1984;85:497-501.  Back to cited text no. 13
    
14.
Materne R, Vranckx P, Pauls C, Coche EE, Deprez P, Van Beers BE. Pancreaticopleural fistula: Diagnosis with magnetic resonance pancreatography. Chest 2000;117:912-4.  Back to cited text no. 14
    


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