CASEREPORTS

PancreaticPseudocystinFederalMedicalCentre,GombeandReviewofLiterature

*OjoEOFMCS **BabayoUD FWACS *DepartmentofSurgery,FederalMedicalCentre,Gombe** DepartmentofSurgery,UniversityofMaiduguri TeachingHospital,Maiduguri

Abstract may be of grave consequence to the patient. This Background: Pseudocyst arises from pancreatic makes prompt diagnosis with a rapid and efficient inflammation or ductal disruption and is the commonest intervention very vital to limiting complications from cystic lesion of the pancreas. Although spontaneous pancreatic pseudocysts. resolution is common particularly in its early phase, progression of the cyst may culminate in grave Case 1. consequences for the patient. A.M was a 27years old, multiparous housewife from The aim of the study is to highlight the presentation, Jalingo who was referred to us with 3months history of management and outcome of pseudocyst in an recurrent exacerbation of epigastric pain which radiated environment where it is reportedly uncommon. to the back and was at presentation associated with Methodology: Case series with review of literature. progressive abdominal distension, easy satiety, Result: Although pancreatic pseudocyst is not very occasional vomiting and weight loss. She had no frequently seen in this environment, it is not entirely rare jaundice. She was previously being managed for but is frequently misdiagnosed. Non specific symptoms in unconfirmed peptic ulcer disease dating back to 7 the early stage and subsequent confusion with various months at a pheripheral hospital and was referred to our intra abdominal pathologies are among the reasons for centre on account of suspected ovarian mass when her late diagnosis and delayed intervention. abdominal distension became gross. Examination Conclusion: Pseudocyst is amenable to surgical revealed a young anicteric woman with PR of 72b/m and treatment. Early recognition is imperative in preventing BP of 130/80mmHg with a non tender, non pulsatile complications and in defining the true incidence of this epigastric mass extending to the pelvic region. disease entity in our locality. Clinical suspicion in every setting of pancreatitis complemented by serum amylase USS showed a 14.4 x 13.4cm low level echo cyst with estimations and early ultrasound scan will improve the multiple gallbladder tiny echogenic foci casting acoustic diagnosis. shadow with normal wall thickness. Barium meal

th showed a soft tissue, fairly rounded mass in the lesser DateAccepted for Publication: 19 April 2010 sac region with normal duodenal cap and C loop. LFT NigerJMed 2010: 223 - Copyright©2010 Nigerian Journal of Medicine was normal and RBS was 4.2mmol/L. A working diagnosis of pancreatic pseudocyst to rule Introduction out mesenteric cyst was made and she had laparatomy Pseudocyst is the commonest cystic lesion of the with findings at operation including: A thick walled, pancreas and mostly complicates acute or chronic unilocular lesser sac cyst containing debris with pancreatitis following alcoholism, cholelithiasis and adhesion to posterior gastric wall, inferior displacement trauma. It differs from a true pancreatic cyst by lack of an of the transverse colon and a gallbladder which epithelial lining and constitutes about two-thirds of all contained multiple tiny stones. She had pancreatic cystic lesions.1 It is reportedly an uncommon cystogastrostomy, excision of cyst wall for biopsy and clinical entity in our environment especially in areas cholecystectomy. Pseudocyst was confirmed at biopsy. where alcoholic beverages are forbidden by religion and Her post operative period was uneventful and she was culture2 but the exact incidence is unknown.3 Pancreatic discharged on post operative day ten. pseudocyst can occur in various locations in the body and mimic pathology in diverse organs but it is typically found Case 2 in the lesser sac.4 Although spontaneous resolution S.A was a 26year old male, petrol hawker, who following a non-operative treatment is common sustained blunt injury to the abdomen when he was particularly in the early stage, progression of the disease knocked down by a vehicle 8 weeks earlier. He was

CorrespondencetoDrOjoEO,P.O.Box1786,Jos,PlateauState,Nigeria,Email: [email protected]

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initially managed in a peripheral hospital for this injury and volume of 684cm.3 The RBS was 8.0mmol/L. He had was subsequently discharged. However, he noticed cystogastrostomy following a diagnosis of pancreatic persistent abdominal pain which became worse 2 weeks pseudocyst which was confirmed at laparatomy. He after his initial hospital discharge and reported back to the developed superficial wound infection and was peripheral hospital where he was managed with discharged home 12days post operation on daily wound analgesics and antacids for peptic ulcer disease. This dressing. was nonetheless followed by progressive abdominal distension with transient, spontaneously regressed Discussion jaundice of 4 days duration. He was referred to our centre Pancreatic pseudocyst is a collection of pancreatic on account of suspected chronic liver disease when he secretions usually rich in digestive enzymes, necrotic developed vomiting, difficulty in lying down/breathing, tissue, old blood which is walled off by fibrous or paedal edema and skin rashes 2 months later. There was granulation tissue.4 Pseudocysts vary in their sizes and no past history of abdominal pain or alcohol ingestion. may be single or multiple. They could be located far from the pancreas (extra-pancreatic) but they are typically USS confirmed the presence of a huge, thick walled, located in the lesser sac (peri-pancreatic) encased by cystic epigastric mass while a barium a meal and follow adjacent viscera such as the stomach, colon, transverse through excluded intragastric or duodenal lesions. His mesocolon, gastrocolic omentum and pancreas.3 This LFT was normal with Alkaline Phosphatase of 153IU/L usually makes mobility exceptional for pseudocysts and and Serum amylase of 41IU/L. An epigastric cystic mass was a cause of clinical confusion where it was reported.5 with a downward bulge through the transverse In this series, all our cases were large unilocular cysts mesocolon was confirmed at laparatomy and he had a located in the lesser sac. Roux en Y Cystojejunostomy done. The postoperative period was uneventful and the histopathology report Pseudocysts are commonly found in the adults due to excluded neoplasm. the incidence of gallstone diseases and alcoholism but could be found in children where it usually follows blunt CASE 3 abdominal injuries.6 The cases in our report were HAA was a 27years old male Fulani driver who loss however all in their third decades of life which may be consciousness and sustained blunt abdominal injury in a due to the small number reported. The age range also road traffic accident when his vehicle loss control and hit a differs from that of five patients reported by Agbakuru et tree. He was transferred to us 2 days later after he had al in Ile-ife over a 10years period whose age ranged regained back his consciousness at the referral hospital from 23 to 70year and this may be due to the inclusion of where he was initially managed. When seen at the A&E patients in whom the aetiology of their cysts was due to he had abdominal pain and distension. He was in pains, alcoholism in their study compared to ours.7 pale, dehydrated but anicteric. His PR and BP were Most pseudocysts are asymptomatic but can manifest 118b/m and 130/80mmHg respectively. Examination with a wide range of clinical problems depending upon revealed distended abdomen with generalized abdominal their location, the extent of the fluid collected or the tenderness maximal in the epigastrium with resonant presence of complications such as infection. percussion notes and hypoactive bowel sounds. An Nevertheless, the earlier symptoms are usually non- assessment of peritonitis from blunt abdominal injury to specific, consisting of epigastric pain, nausea, vomiting, rule out perforated viscus was made and patient had fever or weight loss which may make diagnosis difficult emergency exploratory laparatomy. The intraoperative particularly where the disease is not been suspected.3 A finding were injury to the left lobe of the liver, pancreatic pseudocyst typically forms from seepage of fluid into the injury with zone 2 retroperitoneal haematoma extending lesser sac through the posterior layer of the parietal to around the greater curvature of the stomach, the peritoneum which subsequently results into a well transverse mesocolon and widespread areas of fat circumscribed, sessile epigastric mass immobilized by necrosis. The liver laceration was repaired, peritoneum adhesion to the adjacent structures. Abdominal mass is was lavage with normal saline and the abdomen closed its most common mode of presentation and this may be enmass. associated with pain and tenderness due to However, the patient re-presented 2 months later, with a inflammation or expansion of the cyst. Diagnosis may huge progressive epigastiric mass associated with however be confounded by confusing this resultant discomfort and early satiety.USS revealed an epigastric mass with several pathologies which includes aortic cystic mass measuring 83cm x123cm x 128cm and a aneurysm, panreatic neoplasms, omental/mesenteric

224 PancreaticPseudocystinFederalMedicalCentre: *OjoEO**BabayoUD

cyst and lesser sac tumors8,9 in cases of peri-pancreatic Extra-pancreatic pseudocysts collection at various sites pseudocysts. The situation is more challenging in the is facilitated by proteolytic enzymatic digestion and tropics with inaccessible or few diagnostic facilities in the tracking of fluid along established tissue planes. presence of several disease entities that may mimic the Pseudocysts could be found in the mediastinum or the presentation of pseudocysts10,11 . Two of the patients in this neck when the fluid dissects between the crura of the series were erroneously treated for unconfirmed peptic diaphragm13 . Perirenal cysts may occur when fluid ulcer disease during the early phase of their disease and tracks down from the posterior part or tail of the were also mis-diagnosed and referred as cases of ovarian pancreas through the anterior pararenal space en route mass and chronic liver disease without suspecting the lumbar gutters and may finally end up in the inguinal pancreatic pseudocysts. Early diagnosis of pancreatic region or the scrotum14,19 . Intra parenchymal pseudocysts pseudocyst will always require a high index of clinical accumulation within the solid organs may occur along suspicion particularly in every settings of pancreatitis their vascular or ligamentous attachment as is the case which may be further corroborated by an elevated in splenic and liver parenchyma pseudocysts where the amylase level.Although an elevated serum amylase is not fluid gain access through the splenic hilar vessels and exclusive to pseudocyst, a persistently elevated level the hepatogastric ligament respectively. Intra typically above 1000 IU/L will always suggest the pancreatic pseudocysts are more related to chronic possibility of development of pseudocyst as a pancreatitis and has been reported in heterotopic complication of pancreatitis. Such elevated enzyme level pancreas20,21 . may also arouse early suspicion to search for extra- pancreatic pseudocysts which are formed far away from Ultrasonography is a cheap, widely available, readily the pancreas and diagnosis could then be confirmed by conductible investigative modality with a reasonable demonstrating elevated enzyme level in the aspirates degree of sensitivity and remains the initial radiological investigation of choice. It is useful in defining the size of from the extra-pancreatic collections12,13,14 . Unfortunately, amylase estimation is not quite a regular and routine the cyst, its wall thickness, its relationship to the investigation in most facilities in our locality and only one digestive system, the presence of biliary calculi and of our patients had amylase estimation done. presence of dilatation of the biliary tract or septae within the cyst. It is a valuable tool for postoperative patient The incidence of abdominal pain in pseudocyst reportedly follow up as well as early localisation of pseudocysts. ranges from 76-100%7,15 but featured in all the patients in Endoscopic ultrasonography has the ability to this series. Complications from pancreatic pseudocyst demonstrate the presence of vessels or aneurysms in a can occur due to compression, invasion or rupture of pseudocyst and is of great value in avoiding hazardous pseudocyst into surrounding structures besides infection bleeding during endoscopic cystenterostomy. An upper and hemorrhage16 . The compressive effect on the gastrointestinal endoscopy has the ability to gastrium, duodenum or the biliary tract may lead to early differentiate an intragastric pathology from pseudocyst satiety, gastric outlet obstruction with vomiting or and will also demonstrate extragastric compression obstructive jaundice15 . Vascular compression of the from a large pseudocyst. A barium meal serves as an inferior venal cava, portal or iliac vessels in gross cases alternative investigation in demonstrating intragastric may cause paedal swellings, portal hypertension or pathology where endoscopy is not available and can organomegally and the aorta may be involved in rare define the relationship of the cyst to the upper digestive cases17 . Pancreatic ascites can result from rupture or tract. Chest X ray will confirm the presence of an disruption of the pancreatic duct and there may be pleural accompanying left or bilateral pleural effusion. CT is or pericardial effusion as well as spontaneous infection of invaluable in situations where there is limitation with the cyst. Upper gastrointestinal bleeding may arise from ultrasonography such as in mediastinal pseudocysts vascular erosion into the splenic, gastroduodenal and and for characterising concurrent or underlying pancreaticoduodenal arteries or rupturing of a pancreatic lesions. MRI has the ability to demonstrate pseudoaneurysm. Bleeding into the pancreatic duct very well the presence or extension of fistulous tract into (hemosuccus pancreaticus) may account for the the pancreas and is effective in detecting the presence remaining cases of gastrointestinal haemorrhage. of a solid component to the cyst as well as differentiating Pseudocyst fistulas may occur and the colon, stomach, between organized necrosis and a pseudocyst22 . duodenum and less commonly, the esophagus are the Endoscopic retrograde pancreatography may detect more frequently involved organs with rare biliary tract cysts not seen by ultrasound; demonstrate the involvement18 . presence of communications between a pseudocyst and the pancreatic duct which is of important

NigerianJournalofMedicine, Vol.19,No.2April-June2010,ISSN11152613 225 PancreaticPseudocystinFederalMedicalCentre: *OjoEO**BabayoUD consideration in the choice of treatment as the risk of infeasible in acute pseudocyst which occurs within 3- pancreaticocutaneous fistulae is lower in endoscopic 4weeks of an attack of acute pancreatitis with a wall of drainage than percutaneous methods. It is equally early, immature granulation tissue as oppose to chronic efficient in defining pancreatic ductal pathology (such as pseudocysts which has persisted for more than 6 stricture and obstruction) which may likely prevent weeks and has walls of normal granulation or fibrous spontaneous resolution21 . MRCP has similar sensitivity to tissue1 .Some studies have however demonstrated the ERCP but has the advantage of providing images of the feasibility of anastomosis earlier than 6 weeks30 . The pancreatic ducts in their natural state by not involving the appearance of an isoenzyme of amylase called “old distension of the ducts by the injection of contrast amylase” in serum has been found predictive of cyst media23 .When pancreatography is necessary, it is better wall maturation and was introduced as a biochemical within a day or two of operation under antibiotic cover to marker and timer for performing a safe internal lessen the chance of infection. Angiography is not a drainage21 . routine investigation of pseudocyst but in the setting of pseudoaneurysms resulting from vascular erosion, it The factors guiding the specific drainage site includes: could serve both as a diagnostic and therapeutic size and location of the cyst, condition of its wall, intervention. relation of cyst to the gastrointestine, rapidity of its growth and imminence of rupture , presence of Liver function test, Percutaneous transhepatic complications ( such as rupture, massive suppuration cholangiography and other ancillary investigations may or active haemorrhage of the cyst) and the patients’ complement ultrasonography where there is obstructive age/ general condition.6 Retrogastric anteriorly jaundice. Microbiology will isolate infective organisms enlarging cysts are best drained by a posterior while Cytology or biopsy of the cyst wall will identify cystogastrostomy of Jurasz and those on the neoplastic cysts. Carcinoembryonic antigen (CEA) and pancreatic head abutting on the duodenum by carcinoembryonic antigen-125 (CEA-125) tumor marker cystoduodenostomy of Kerschner29 . Large cysts, with levels are low in pseudocysts and elevated in tumors. A inferior enlargement, bulging through the transverse carcinoembryonic antigen (CEA) level greater than 400 mesocolon especially when they are neither adherent ng/mL within the cyst fluid strongly suggests malignancy. to stomach nor duodenum are best drained by Roux- An elevated serum glucose level may herald the en-Y cystojejunostomy2 9 . The prerequisite for development of diabetes mellitus post pancreatic performing a cystoenterostomies include the absence inflammation. of intra cyst papillary growth, pseudoaneurysm or The initially approach to treatment of pseudocysts is compartments of the cyst and absent epithelial lining of 26 usually conservative as cysts may spontaneously resolve the cyst wall where a frozen section is carried out . when small (less than 6cm in size), are in the acute phase Cystogastrostomy and Roux-en-Y cystojenostomy (less than six weeks), have a wall thickness not more than were employed in treating our patients with satisfactory 1cm and when there are no other pancreatic duct outcomes. abnormality other than the pancreatic duct-pseudocyst Endoscopic drainage is a more recent form of internal communication24 . Up to 50% of pseudocysts may regress drainage with all the full compliments of a minimally on expectant management25 . The surgical approach to invasive procedure. It entails an image guided widening pancreatic pseudocyst entails drainage of the cyst using to about 2cm of a diathermic puncture made between various techniques with considerations given to the the digestive tract mucosal and the cyst followed by anatomy of the cyst, the pathology present and the insertion of nasocystic catheter for a few days. general condition of the patient26 . Although medical Endoscopic drainage may be achieved by management has been tried and anecdotally reported to transpapillary stenting when the pseudocyst be successful, the therapy is usually prolonged27 communicates with the pancreatic duct or by transmural gastric/duodenal wall drainage via the site Internal drainage is the traditionally preferred method of of maximal extrinsic compression and visible treatment and involves an anastomosis between the cyst endoscopic ‘bulge’31,32. Endoscopic drainage has also and the upper digestive tract (stomach, duodenum or been extended to extra pancreatic pseudocysts as in jejunum). It has been accomplished by open, endoscopic mediastinal and intra-hepatic pseudocysts1 3 , 3 2 , 3 3 . or laparoscopic techniques28,29 . Success of internal Transmural endoscopic cystoduodenostomy and drainage depends on the presence of a mature cyst wall to cystojejunostomy are more technically infeasible when hold the stitches in the open technique and this may be the distance between the viscera and the cyst is more

NigerianJournalofMedicine, Vol.19,No.2April-June2010,ISSN11152613 226 PancreaticPseudocystinFederalMedicalCentre: *OjoEO**BabayoUD than 1 cm and are hazardous in the presence of pseudocysts. Here, duct-pseudocyst communication is pseudoaneurysm or intervening cyst wall vessels which often present and the pancreatic duct is diseased but serves as contraindications to the procedures34,35 . The not strictured. Surgical internal drainage is usually a recurrence rate for all endoscopic procedures for the successful treatment although percutaneous drainage treatment of pancreatic pseudocysts is less than 5% and may be possible, but may have to be prolonged. Group the complication rate is approximately10%36 . The possible III patients are those with chronic ‘retention’ complications from this procedure include serious pseudocysts who has grossly diseased and strictured bleeding from unsuspected pseudoaneurysm or pancreatic duct in the presence of duct-pseudocyst intervening vessels in the cyst wall, pseudocyst infection communication. In this category, cure demands and bowel perforation. The drawbacks to transpapillary operative procedures that address the specific stenting particularly include exacerbation of pancreatitis, underlying ductal pathology and surgical internal stent occlusion leading to recurrent duct blow-out and drainage has a high recurrence rate while percutaneous pseudocyst formation or chronic pancreatitis37 . drainage is contraindicated. External drainage provides a rescue treatment where Haemorrhage is the single most important complication there is a rapidly growing cyst with immature wall and of pseudocyst contributing up to 50% mortality in some imminent rupture especially in poor risk/ critically ill series3 0 . Managing a pseudocyst with active patients.6 It involves the drainage of the cyst outside the haemorrhage could be a formidable threat but could be abdominal cavity either by open or Image guided achieved by vascular embolisation prior to surgery, percutaneous catheter. Sequela of this form of treatment balloon catheterization, suture ligation of the vessel or includes infection, fistula formation, skin enzymatic by pancreatic resection together with the pseudocyst digestion, bowel catheter erosion in prolonged drainage where the haemorrhage is uncontrollable. Equally and frequent cyst recurrence38 . It should consequently not amenable to pancreatic resections are pseudocysts be undertaken in the presence of pancreatic necrosis or a located at the pancreatic tail6 . solid non-drainable pancreatic mass, lack of safe access Pancreaticoduodenectomy may suffice where a co- route, active pseudocyst hemorrhage or compartments existing pancreatic tumor is discovered or in within the cyst. Serial ultrasound monitoring should be multiloculated cyst unyielding to other form of drainage. incorporated as part of treatment to ascertain the efficacy However, this could be a daunting operation with a high of the drainage. Sandostatin, a “somatostatin analogue” morbidity and mortality and is best left to a skilled which is noted for decreasing the basal and stimulated surgeon in pancreatic surgery. Pancreatic pseudocysts pancreatic secretion has been used as an adjuvant to complicated by biliary problems such as gallstones or catheter drainage to improve its efficacy3 9 . unresolved ductal compression after drainage of the Marsupialization is now rarely undertaken as a form of cyst are treated on their own merits besides the drainage except where the content is massively infected26 . drainage of the pseudocyst. We as a result carried out cholecystectomy in one of our patients with associated Complications reported with percutaneous drainage gallbladder stone. The mortality of pancreatic include infection, pneumothorax, pleural effusion, and pseudocyst is increased in the presence of minor haemorrhage24 . Image guidance percutaneous complications which include: Pancreatic necrosis or catheter drainage is associated with success rate of 90 to abscess, haemorrhage, rupture into cavities and its 95% but simple aspiration of pseudocyst is associated sequela, intestinal obstruction and fistula formation. with recurrence rate of 70% and should be Therefore, prompt drainage of the cyst by the most discouraged24,25,40 . appropriate mean once spontaneous resolution is D’Egidio et al has earlier emphasised the need to classify unlikely as seen in the cases presented in this study pancreatic pseudocyst to enable the adoption of the most remains vital to ensuring a good outcome by eschewing appropriate form of treatment and evolved a clinico- grievous consequences. radiographic pseudocyst classification38 .This comprises In conclusion, although pancreatic pseudocyst is of patients with acute, ‘post-necrotic’ pseudocysts having uncommonly seen in our locality, it is not an entirely rare a normal pancreatic duct anatomy and rarely duct- pathology but is frequently misdiagnosed with delayed pseudocyst communication in Group I category. Here, intervention. The symptoms are non specific for early Percutaneous drainage is curative in all patients when lesions causing confusion with variable pathologies used. Group II patients include those already suffering such as peptic ulcer disease. Furthermore, from chronic pancreatitis that develops ‘post-necrotic’ corroborative diagnostic facilities such as amylase

NigerianJournalofMedicine, Vol.19,No.2April-June2010,ISSN11152613 227 PancreaticPseudocystinFederalMedicalCentre: *OjoEO**BabayoUD estimation may not be readily accessed. The diagnosis of detecting those cysts that may spontaneously regress pancreatic pseudocysts needs to always be suspected, and not likely be reckoned with when estimating the then investigated and appropriately followed up. The incidence of the disease. In our setting, internal practice of treating peptic ulcer disease without drainage provides satisfactory outcome of treatment endoscopic confirmation in our environment should be but is mainly in form of open anastomosis between cyst reviewed. Epigastric pain and swelling being among the and the upper digestive tract as there is limited more definite features of peri-pancreatic pseudocyst experience with endoscopic drainage in this part of the should always point to the possibility of pseudocyst when world. Endoscopy in form of ERCP and MRCP are they occur and be followed by an ultrasound scan for a invaluable for unravelling the underlying ductal prompt diagnosis. pathology and adequate categorisation of pseudocysts. While late presentations are fraught with grievous Acquisition of relevant minimally invasive/endoscopic consequences, early diagnosis apart from preventing technology and expertise will therefore allow us adopt complications will be useful in ascertaining the true this approach where it is of superior advantage. incidence of this disease entity in our environment by

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