A rare case of pancreaticopleural fistula patient presented in surgery OPD

Pancreatico-pleural fistula is rare and infrequent complication of commonly occurring chronic pancreatitis leading to an extra-peritoneal abnormal connection between the pancreatic system and pleural cavity. Diagnosis needs high-level clinical suspicion to avoid delay in the diagnosis as the patient presents with respiratory distress rather than any abdominal symptom and produces large quantities of pleural fluid intractable of pleural tapping or chest drain. Diagnosis of the fistula is clicked by elevated pleural fluid amylase. Various imaging options are available with their unique importance like CECT, ERCP and MRCP. In a low resource, setup CECT becomes a useful modality to delineate the pancreatic parenchymal changes, pancreatic duct anatomy and fluid collection, thus aid in the diagnosis. Treatment modalities depending on structural anatomy of the duct and parenchymal destruction are either Medical, Conservative and Surgical. Here our patient presented with massive left sided pleural effusion resistant to surgical intervention secondary to chronic pancreatitis in a 28-year man later diagnosed as Pancreatico-pleural fistula on CECT. The patient underwent distal pancreatectomy with splenectomy with decortication of the lung with excision of PPF. The patient now is continuous follow-up for chronic pancreatitis and is symptom-free from last 2 years.

Pancreatico-pleural istula, chronic pancreatitis, elevated pleural luid amylase A Pancreatico-pleural istula is rare and infrequent complication of commonly occurring chronic pancreatitis leading to an extra-peritoneal abnormal connection between the pancreatic system and pleural cavity. Diagnosis needs high-level clinical suspicion to avoid delay in the diagnosis as the patient presents with respiratory distress rather than any abdominal symptom and produces large quantities of pleural luid intractable of pleural tapping or chest drain. Diagnosis of the istula is clicked by elevated pleural luid amylase. Various imaging options are available with their unique importance like CECT, ERCP and MRCP. In a low resource, setup CECT becomes a useful modality to delineate the pancreatic parenchymal changes, pancreatic duct anatomy and luid collection, thus aid in the diagnosis. Treatment modalities depending on structural anatomy of the duct and parenchymal destruction are either Medical, Conservative and Surgical. Here our patient presented with massive left sided pleural effusion resistant to surgical intervention secondary to chronic pancreatitis in a 28-year man later diagnosed as Pancreatico-pleural istula on CECT. The patient underwent distal pancreatectomy with splenectomy with decortication of the lung with excision of PPF. The patient now is continuous follow-up for chronic pancreatitis and is symptom-free from last 2 years.

INTRODUCTION
PPF is infrequent complication rarely in acute or commonly in chronic-pancreatitis caused by in lammatory/traumatic injury to pancreatic-duct. Due to pancreatic in lammation, istula is formed between PD and pleural-space, leading to pancreatic secretion draining into the pleural cavity, causing enormous pleural-effusion high in amylase. Incidence of less than 0.4% of pleural effusion due to CP. The total number of cases reported till 2017 of PPF worldwide is 40 (Rao and Raghavendra, 2015;Ramahi et al., 2019).
Patients present with pulmonary symptoms like respiratory-distress, cough with expectoration due to pleural-effusion rather than pain in the abdomen due to pancreatitis (Ali et al., 2009). Most PPF's are formed by the leak of pancreatic secretion from posterior distortion of PD or posterior rupture of pancreatic pseudocyst. Once the diagnosis is con irmed, treatment is usually directed to obliterate istula between the pancreas and pleural cavity conservatively, endoscopically or surgically (Ramahi et al., 2019;Tay and Chang, 2013).

CASE REPORT
28-year male, chronic alcoholic and smoker since past 14 years, presented surgery with intercostaldrain with massive secretion and had breathlessness since 7 days, dull-aching epigastric pain since 9 months, cough with expectoration since 2 months. The patient had a history of excessive consumption of alcohol 8 days ago, after which he experienced the aggravation of symptoms. The patient was diagnosed case of pancreatitis associated massive recurrent pleural-effusion for which he was referred to our centre.
The patient was vitally stable with normal oxygen saturation but had reduced air entry in the basal area of the left lung with ICD in situ on the left side with 200 ml pleural luid collected within it. Per abdominal examination revealed tenderness in the epigastric region with no guarding/ rigidity or distension with normal bowel sounds.
All biochemical investigations were normal except serum Amylase and lipase, which increased i.e.556 and 260u/l. Chest x-ray showed left-sided pleural-effusion with ICD in 5 th intercostal-space.
Diagnosis of Acute-on-chronic pancreatitis with left sided pleural-effusion with suspicion of PPF, pleural-luid was subjected to estimation of amylase and lipase that was very high, i.e. 32,164 u/l and 55,670u/l respectively.
On CECT thorax/abdomen, there was moderate pleural effusion associated collapse of the left lung, Acute-on-chronic pancreatitis with multiple pseudocysts in body and tail in sub-diaphragmatic and paracolic region with normal pancreatic-duct, pseudo-aneurysm of size 10x9 mm from splenic artery in relation to pseudo-pancreatic cyst. A connection between pleura and pancreas was clearly visible hence de initive surgery was conducted on this patient (Figure 1).
Prior to surgery, the patient underwent gel foam embolization of splenic artery to limit bleeding; de initive surgery was performed in the form of distal pancreatectomy with splenectomy along with excision of all pseudocysts and excision of the istulous tract by thoraco-abdominal incision. Decortication was required to release ibrosis for complete expansion of the lung (Figure 2 ).
Post-operatively patient was administered with somatostatin-analogue (octreotide) for 3days, assessment of chest drain showed declining fashion of amylase. Patient recovery postoperatively was uneventful followed-by ICD removal and now is followed up for chronic-pancreatitis (Figure 3). PPF is rarely found in acute/chronic pancreatitis/surgical/traumatic pancreatic disruption. The common etiological factor is chronic alcoholic pancreatitis with pseudocyst in adults and Biliary duct obstruction in children others include gallstones, abdominal trauma and pancreatic-duct anomalies (Aswani and Hira, 2015).

DISCUSSION
Chronic pancreatitis causing PPF is accelerated unchangeable in lammatory activity portrayed by substitution of normal pancreatic parenchyma with

Figure 3: Arrow depicting pancreatico-pleural connection
ibrotic connective-tissue after repetitive attacks of in lammation leading to extravasation of in lammatory luid forming granulated istulous tract between pancreas and Pleural-cavity (Ali et al., 2009;Aswani and Hira, 2015). A posterior distortion of PD or posterior pseudo-pancreatic cyst blowout into retro-peritoneum results into the pancreatic section to ascend to pleural space through natural ori ices like aortic or hiatus or directly through diaphragm on either side, rarely if distortion is anterior resultant is Pancreatico-peritoneal istula giving rise to ascites (Ramahi et al., 2019;Lee et al., 2014).
Pleural-effusion secondary to PPF and reactive pleural-effusion in chronic pancreatitis are discriminated from each other by a high level of amylase (>1000u/l), Clinical picture demonstrates a middle aged 30-40-year male, with history of excessive alcoholism with dominant respiratory symptoms (Lee et al., 2014). Manifestations include dyspnoea (65%), abdominal pain (29%), cough (27%), chest pain, fever, back pain, haemoptysis, fatigue or orthopnoea. Patient present with recurrent pleuraleffusion otherwise resistant to surgical treatment and is rapid in accumulating (Rao and Raghavendra, 2015).
Radiological techniques to delineate istula in order of sensitivity and availability are CECT (79%), MRCP (80%) and ERCP (46 to 78%). Ultrasound is done but does not help in diagnosis since bowel gas overlaps the upper-abdomen (Tay and Chang, 2013). CT is better since it describes acute/chronic changes with parenchymal/ductal changes and luid collec-tion of the pancreas (Sonoda et al., 2012). ERCP is better than both as to its diagnostic and therapeutic providing description about istula, ampulla and ductal anatomy through invasive needs experienced gastroenterologist to perform and fails if the istula is located beyond the site of obstruction. MRCP is astonishing since help in the diagnosis of site of istula beyond the obstruction, de ines pancreatic parenchyma and strati ies management protocols, also is non-invasive thus making it investigation-ofchoice, also no use of contrast media makes it nonallergic with no risk of infection (Rao and Raghavendra, 2015;Aswani and Hira, 2015).

3) Surgical
Management based on ductal anatomy on MRCP is described as, 1. Medical management with somatostatin analogues with thoracentesis/chest drain in a patient with normal or mild dilatation of PD.
2. ERCP stenting in patients with PD disruption in head or body of pancreas or distal stricture and 3. Surgical management in the form of pancreatic resection or pancreatico-enteric anastomosis in patients with complete PD disruption, complete stricture of PD, PD obstruction proximal to istula and noncompliance with medical and endoscopic treatment (Aswani and Hira, 2015).
Conservative management with somatostatin analogues (Octreotide) with adjunct pleural tapping or ICD insertion along with rest to bowels, gives time to istula to resolve and thus allowing PE to settle down by reducing pancreatic secretion by its gastric inhibitory effect and istula output. The success rate is 31-65% within 2-3weeks, and failure to which need endoscopic or surgical treatment (Ramahi et al., 2019;Sonoda et al., 2012).
ERCP-stenting to PD is a valuable option as it spares patient to undergo major surgical-stress working by mechanically blocking abnormal connection of PD with pleura and keeping PD open and obstructionfree, so secretions pass to duodenum thus escaping abnormal pleural connection (Ramahi et al., 2019;Aswani and Hira, 2015). Success-rate is variable reported by Khan (100%), Pai (96.4%) and Varadarajulu (55%) (Sonoda et al., 2012).
The main principle of surgical management is in the form of free drainage of pancreatic secretion by pancreatico-enteric connection with or without pancreatic resections, excision of tract and pseudocyst. Advanced surgical treatment has low morbidity and mortality associated with quicker recovery and better outcomes than medical or endoscopic treatment. Surgical management has proved better and more de initive as the patient takes lesser time to recover from post-surgical stress in the postoperative phase as compared to the recovery of conservative and endoscopic management. Pancreatic parenchymal preservation is particularly important as the majority of patients with PPF have diminished reserves of endocrine or exocrine functions due to chronic pancreatitis (Ramahi et al., 2019;Sonoda et al., 2012). The success rate of surgical management was found to be highest (94%), whereas the lowest for medical therapy (33%) (Aswani and Hira, 2015).

CONCLUSIONS
Diagnosis of PPF is based on high clinical suspicion due to recurrent massive pleural effusion refractory to ICD with markedly increased PF amylase and radio-imaging features. Although an initial medical and endoscopic management is recommended; still surgical management remains the de initive modality of choice for PPF as it is associated with chronic, recurrent pancreatitis with frequent relapses but treatment differs according to the clinical status of the patient, PD anatomy, pseudocysts and other related complications of CP.