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CASE REPORT

Management of a Complicated Pancreatic : Report of a Case and Review of the Literature

Apostolos Αngelis, Stylianos Kykalos, Zoe Garoufalia, Elli Karatza, Nikolaos Garmpis, Christos Damaskos, Theodore Karatzas

Second Department of Propedeutic Surgery, Laiko General Hospital, Medical School, National and Kapodistrian University of Athens, Greece

ABSTRACT Case report Herein we present a case of a huge complicated pancreatic pseudocyst following an episode of acute necrotizing , the treatment approaches and a short review of the literature. Methods An on-line survey in PubMed was conducted using the terms the patient regarding his treatment. Results Treatment of a pancreatic pseudocyst is a challenging process requiring a close follow-up of the“acute patient pancreatitis-complications”, and a fully equipped institution “pancreatic with pseudocyst” medical team and ready “pancreatic to cope against pseudocyst any possible treatment”. complications We also used of the the different data from therapeutic the file of interventions including open surgery. Conclusion Pancreatic pseudocyst is a of acute or which should be managed closely for a longstanding period and treated effectively by endoscopic and surgical means, either alone or in combination, so that the outcome of this complication to be in favor of the patient.

INTRODUCTION In this case we present a patient with a huge pancreatic pseudocyst complicated after endoscopic drainage episodes of pancreatitis that result in tissue a short review of the literature of the different treatment or Pancreaticdisruption pseudocystof a pancreatic is a fluid-filled duct. According cavity after to acute the strategiesprocedures of and the finallypancreatic resolved . by open surgery, and also CASE REPORT developsRevised Atlanta an enhancing classification, capsule as earliera pseudocyst than fourshould week be A sixty-two-years-old man was referred to our hospital, aftercharacterized onset of acuteevery pancreatitis. acute pancreatic The communication fluid collection withthat 5 months after an episode of severe acute necrotizing the pancreatic ductal system is initially always present pancreatitis in a district hospital. The patient was and may further remain or seal off spontaneously during diagnosed with pancreatic pseudocyst, three weeks after the clinical course [1]. Most pseudocysts present minor the remission of the episode. The pseudocyst was 18 cm in symptoms and are uncomplicated. The vast majority of diameter and was causing restrictive symptoms, mostly of pseudocysts (less than 6 cm) have thin wall and usually early satiety and loss of weight (15 kg) during his hospital resolve spontaneously. Large pseudocysts are often in stay. The patient’s history includes goiter, hyperlipidemia continuity with the pancreas, and thick-walled rarely and chronic obstructive pulmonary disease. communicate with the pancreatic ductal system [2]. On CT scan with pros and IV contrast agent there was Most large pancreatic pseudocysts are likely to remain requiring intervention only in the presence of complications (, , splenic vein thrombosis areafound of a the large lesser fluid sac. collection Also, there of 18was cm found in diameter a pancreatic with etc.), and obstructive symptoms of , , or necrosisdebris (hemorrhagic of 70-80%. fluid and necrotic material) in the stomach. The most common complaints of the patients are early satiety, and after meals [3]. In our hospital, the patient continued to present symptoms of early satiety and epigastric discomfort Received March 24th, 2018 - Accepted May 29th, 2018 after meals and was severely debilitated. On a second CT Keywords Case Management; Pancreatic Pseudocyst; Pancreatitis Correspondence Theodore Karatzas (Figure 1) Second Department of Propedeutic Surgery scan the pancreatic fluid collection was further enlarged Laiko General Hospital, Medical School endoscopic drainage of the pseudocyst, so the patient was National and Kapodistrian University of Athens referred to another. As a first-linehospital for treatment interventional was decided endoscopy. the 17 Agiou Thoma Street, 11527, Athens, Greece The endoscopic EUS showed a cystic mass behind the Tel +30 2107462553 Fax +30 2132061772 stomach pressing its lumen with a large amount of debris E-mail [email protected] inside. Drainage of the pseudocyst through the stomach

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Figure 1. Abdominal CT scan showing a huge (18 cm of diameter) chronic lobulated pancreatic pseudocyst located mainly on the body and tail of the pancreas, surrounded by a thick, dense wall and containing debris with necrotic materialpancreas within was its healthy.fluid-filled Thecavity. patient started having normal material (debris), and a metallic stent was inserted into meals and he was discharged from the hospital in good thewas performed cavity. A withnasogastric 500cc oftube necrotic was placed fluid in and order solid to clinical status. Ten months later the patient remains in avoid aspiration. very good conditions. The patient returned back to our department. The DISCUSSION next day the patient was febrile. The site of infection was attributed to aspiration because of inadequate drainage collections surrounded by a wall composed of collagen of the pancreatic pseudocyst. Another attempt, for more andPancreatic granulation tissue pseudocysts without . are well The defined prevalence fluid has been reported to range from 6% to 18% in the acute EUS. During this new examination there was found that the and from 20% to 40% in chronic pancreatitis respectively. stentefficient remained drainage in placeof the but pseudocyst the debris was of the made, pseudocyst with a newwas Pseudocysts may be drained using a variety of approaches not fully drained. Attempts were made to remove the necrotic or a combination of techniques, especially if an internal material with a basket. A nasogastric tube was inserted drainage is feasible. The decision making concerning directly into the cavity of the pseudocyst aiming to wash out treatment indication and optimal approach is quite the cavity with continuous lavage with normal saline, which eventually would shrink in second phase (Figure 2). accurately the peripancreatic collections accompanying or The symptoms, however, continued and the patient followingchallenging. an Theacute Revised pancreatitis Atlanta [4]. classification categorizes remained highly febrile. Because the clinical status of the patient was getting worse and he became septic it was early in the course of (without cystic decided to take him in the operating room for open surgery wall)Acute and/or pancreatic necrosis fluid may collection or may not(APFC) be present. that develops Acute and drainage of the pseudocyst through the stomach. At necrotic collection (ANC) is the development of necrotizing laparotomy there was found a huge pancreatic pseudocyst strictly attached to the posterior wall of the stomach and material. The distinction between those two conditions to surrounding tissues. Through a posterior gastrotomy an pancreatitis which contains both fluid and necrotizing opening of the pseudocyst was performed and was drained CT images in ANC become more complex. Pancreatic is difficult in it he first week. As the disease progresses debris inside. Also, an extensive necrosectomy of the body without necrosis and form after 4 weeks. Walled-off a large amount of infected pancreatic fluid with necrotic and tail of the pancreas was performed (Figure 3), followed necrosispseudocysts (WON) which is another are encapsulatedcondition which fluid usually collections appears by a large anastomosis between the pseudocyst and the after 4 weeks of the ANC episode and develops a thickened stomach. Post-operatively the patient’s clinical status was nonepithelialized wall between the necrosis and the adjacent improved rapidly and his recovery was uneventful. His tissue. Like ANC, WON may involve pancreatic parenchymal symptoms regressed and he remained afebrile. Repeated tissue. The distinction between WON and pancreatic CT scan showed that pseudocyst regressed on imaging, the pseudocyst is very important as a pseudocyst can be treated necrotic pancreatic bed excised was clean and the remnant

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Figure 2. Endoscopic image of the stent in the stomach: transgastric stenting using endoscopic approach into the pseudocyst cavity with a naso-gastric tube inserted through the cysto-gastrostomy transmural stenting. a b

Figure 3. Open surgical procedure. (a). Transgastric drainage of the pseudocyst; (b). Removal of extensive necrotic tissue from the body and tail of the pancreas.

All these entities can be distinguished by patient’s that helps the clinician to distinguish the characteristics of history, imaging studies or even biochemical analysis of needle aspirate if necessary. cytology and estimation of tumor markers may help towardsa pseudocyst. distinguishing Aspiration cysticof fluid malignancies content with fromconcomitant simple Diagnostic Evaluation pseudocysts. The initial diagnosis of a pancreatic pseudocyst can The clinical course of a pseudocyst highly depends on its size and the time that has elapsed since the diagnosis. ultrasonography. CT scan is often the method of choice caused by an episode of acute pancreatitis, measured forbe establishing initially set the upon diagnosis, the findings with sensitivity of transabdominal up to 100% to have a diameter of up to 4 cm, regress spontaneously and require no treatment, given that they remain and specificity of 98%. EUS is another imaging method JOP. Journal of the Pancreas - http://pancreas.imedpub.com/ - Vol. 19 No. 3 – May 2018. [ISSN 1590-8577] 159 JOP. J Pancreas (Online) 2018 May 33; 19(3):157-163. asymptomatic. On the other hand, chronic pancreatic If technically feasible, endoscopic cystgastrostomy for pseudocysts show a low regression’s rate (less than 10%). pseudocysts is as effective as surgical cystgastrostomy, but with shorter hospital stays, lower costs and higher Indications of Treatment patient physical and mental health scores [7]. However In general, every symptomatic pancreatic pseudocyst other authors as Melman and Gurusamy report that the that has been present for more than six weeks and has not endoscopic approach is inferior in terms of primary regressed under conservative treatment should be treated success in comparison to surgical approach as additional [5]. interventions were needed in the endoscopic group. As Among the possible symptoms and complications, controversial as far as the efficacy of the two approaches the existing literature is conflicting the results are still of a WON, the endoscopic debridement should ideally duodenumthere are may some lead specific to symptomatic situations obstruction, that should while be goal[8, 9]. to Furthermore, revealing and after removing gaining thean accessgranulation in the cavitytissue compressionhighlighted. Firstly, of the commona firm compression bile duct may of the cause stomach stenosis or covering the pseudocyst wall. Regarding the endoscopic and symptomatic . A possible compression of treatment of pseudocysts and WONs, following major vessels may manifest with angina intestinalis, occult recommendations have been stated through several intestinal bleeding due to ischemia, impaired intestinal the gastric or duodenal wall should ideally be less than infection or hemorrhage into the cyst or even formation authors. Firstly, the distance between the pseudocyst and motility and serum lactate elevation. Furthermore, 1 cm, in order to facilitate an effective stent placement course of the pseudocyst. The indication of treatment chosen approach should be through the site of greatest of aany pancreaticopleural asymptomatic pseudocyst fistula may withcomplicate diameter the greaterclinical impression[10]. Furthermore, by the inpseudocyst the absence on ofthe EUS adjacent guidance, gastric the than 4 cm that does not resolve after the period of 6 or duodenal wall [11, 12]. The greater success rates of weeks is relative. The supporters of such an approach endoscopic therapy are noted in cases of single, large claim that these cysts do have an increased possibility for (>5 cm), mature cysts that do not communicate with the complications in the future. A further relative indication pancreatic duct [13]. is the chronic pancreatitis in presence of abnormalities or stones in pancreatic dust. Pseudocysts that correlate It should be mentioned that the presence of malignancy or the formation of pseudoaneurysm should be ruled out, spontaneous regression. before any endoscopic attempt. Especially in the second to these conditions, show significantly lower rate of condition, every endoscopic maneuver may lead to Treatment Options troublesome bleeding. Management of pancreatic pseudocysts include The next question that should be answered is which conservative treatment (watchful monitoring), surgical type of stent should be used. Some authors favor the use drainage (open or laparoscopic), or endoscopic drainage. of pigtail catheters in contrary to straight stents, because In endoscopic drainage, a stent is inserted in order to their complication rate is markedly lower. Moreover, the achieve a connection between the pseudocyst and the placement of multiple wide stents raises the success rate of stomach (usually) or even the proximal part of the small endoscopic intervention, without affecting the morbidity bowel. The stent placement may take place under EUS or mortality [14]. guidance, while an additional insertion of a nasocystic tube (through the stent) may be performed. The optimal Traditional self-expanding metal stents (SEMS) are treatment of pancreatic pseudocysts remains unclear, as designed to anchor in place in a stricture; however, when the data extracted from current literature are inconclusive. Hookey et al. supported that the treatment of pancreatic used for treatment of pancreatic fluid collection, there pseudocysts always necessitates an interdisciplinary is a significant migration risk, as the size and shape of approach [6]. designed for management of pancreatic collections [15]. Muchavailable promising biliary andis the esophageal introduction SEMS of the are so not called specifically lumen- It should be noted that, prior to any intervention, apposing metal stents (LAMS), which make the transmural the pancreatic ductal system should always be initially intervention more effective [16]. This kind of stent ensures investigated. If the pancreatic pseudocyst does have a a larger lumen for drainage and endocystic access, while connection to Wirsung's duct, the transpapillary stent insertion for internal drainage represents the preferable treatment option. The main principle is that any pancreatic wasancors published firmly in in 2012 both and lumens since (intestinalthen LAMS anddo become cystic) ductal disruption should be bridged with endoscopic steadilyshowing more low migrationpopular in rates.endoscopic The first cystgastrostomy referral to LAMS and stenting, because a remaining disruption of pancreatic pancreatic necrosectomy interventions [17, 18]. The safety duct decreases the rate of cyst resolution after drainage. In cases where the transpapillary drainage of the pseudocyst [19, 20]. Their application instead of double pigtail stents is not feasible, the endoscopic approach represents the and efficacy of LAMS is supported by several recent studies alternative to surgical procedures. is strong published evidence that LAMS are associated (DPS) gains constantly field in drainage of WON. There

JOP. Journal of the Pancreas - http://pancreas.imedpub.com/ - Vol. 19 No. 3 – May 2018. [ISSN 1590-8577] 160 JOP. J Pancreas (Online) 2018 May 33; 19(3):157-163. with higher regression rate, fewer needed endoscopic The laparoscopic approach is not always feasible and interventions and shorter hospital stay [21, 22, 23, 24]. requires a skilled surgical team. The drainage principles remain the same as in the open procedure. The reported Despite the existing enthusiasm, we should be aware success rate is 90% with 0% mortality and 9% complications of all possible complications that may accompany the application of endoscopic stents (Pigtails, but mostly SEMS and LAMS). retentionsrate. Finally, or transcutaneousinfected cysts. The drainage recurrence is indicated rate ranges only as an emergency intervention for symptomatic acute fluid or even delayed bleeding (due to stent related erosion) may complication [32]. More specific acute bleeding during drainage procedure high as 70% and percutaneous fistula is a very common In general, surgical (including minimally invasive) of wide metal stents, the presence of pseudoaneurysm or techniques are hard to compare because of an evident varices,occur. Factors and the predisposing blind puction to without bleeding EUS are guidance the placement as well. selection bias. Patients that require transcutaneous intervention are candidates with high morbidity, thus not control, especially if the injured site is located in the cystic eligible for surgical treatment. cavity.The bleeding In exceptional may be cases, quite an troublesome emergency andangiography difficult or to even laparotomy may be indicated. Gurusamy et al. compared four randomized control trials with a total sample of 177 participants, and analyzed Migration of the stent represents a further potential the treatment options of pancreatic pseudocysts, extracting complication. A stent dislocation into the cyst cavity can interesting results. The different treatments included be more problematic, as the cystgastrostomy tract may endoscopic drainage (without EUS guidance), EUS-guided partially or completely close. An unusual complication of drainage, EUS-guided drainage with nasocystic drainage cardia occlusion after stent migration has been recently and open surgical drainage. The authors mention that reported [25]. the overall quality of evidence was very low for all the outcomes, because the trials included a limited number occlusion, predisposing to cyst retention, enlargement of participants and additionally were at high risk of bias. Finally, mucosa growth over the stent may lead to its The analysis of data referring to mortality and serious become quite challenging. Other causes of stent occlusion adverse events showed comparable results between the andor even drainage infection. impairment Furthermore, may be the the interposition stent removal of food may various treatments strategies. On the other hand, the debris or cyst contents, requiring additional endoscopic short-term health-related quality of life (up to 3 months) interventions [26]. was worse and the costs were higher in the open surgical In terms of all possible complications, the administration drainage group than in the EUS-guided drainage group. The EUS-guided drainage with additional nasocystic of SEMS or LAMS should be timely limited. DeSimone et drainage caused fewer adverse events than EUS-guided al. suggest early removal of LAMS, immediately after cyst regression. If long term drainage is needed, such as in cases or endoscopic drainage, and had shorter hospital stay of recurrent pseudocysts, plastic stents or pig tails should be preferred [27]. of endoscopic drainage showed the higher incidence of additionalwhen compared invasive to procedures other modalities. needed Finally,for cyst resolution the group The complication rates after drainage of infected [33]. pseudocysts vary among the published studies. Although few studies present high complication rates after drainage According to the Asian consensus statement on of abscesses or WONs [28, 29], some others report contrary endoscopic management of WON, conservative treatment should represent the initial approach to these patients (watchful waiting approach) [34]. The supportive therapy findings [30, 31]. consists in administration of systemic antibiotics and treatment modality, in cases where all other approaches nutritional support, as well as organ and system support. The role of surgery is clear, representing the definitive The use of endoscopy is preserved for the patients who fail successful operation of pancreatic pseudocyst drainage to improve, despite the aggressive medical treatment of wasare provendescribed to beby notBozeman feasible in or 1882. inadequate. Technically The firstthe pseudocyst usually is incised and debriefed and the drainage procedures are inadequate, necrosectomy should cystic wall is anastomosed wherever possible (stomach, besymptomatic performed. or If infected an endoscopic WON. Finally, approach in cases is not where feasible, the duodenum, small bowel). a surgical necrosectomy (open or laparoscopic) should The rate of surgical success ranges from 90 to 100 %, be undertaken. The authors comment that additional while the average mortality and morbidity are 2.5 % and nasocystic drainage has not been proven to be helpful, so 16 % respectively. The recurrence rate ranges from 0-12 its placement is not recommended. % over the next 5 years, depending mostly on the site CONCLUSION of the pseudocyst and the underlying illness. Moreover, the surgical treatment of pseudocysts due to chronic The treatment of pancreatic pseudocysts should always pancreatitis carries a markedly lower mortality and involve an interdisciplinary therapeutic approach ensuring morbidity. an initial adequate support. Endoscopic interventions,

JOP. Journal of the Pancreas - http://pancreas.imedpub.com/ - Vol. 19 No. 3 – May 2018. [ISSN 1590-8577] 161 JOP. J Pancreas (Online) 2018 May 33; 19(3):157-163. compared to surgical or radiologic approaches are more 14. Giovannini M. What is the best endoscopic treatment for pancreatic often successful when multiple wide stents are placed, and pseudocysts? Gastrointest Endosc 2007; 65: 620-3. [PMID: 17383458] this does not elevate either morbitity or mortality. This 15. is particularly true with regard to placement of pigtail Shah RJ, Shah JN, Waxman I, Kowalski TE, Sanchez-Yague A, Nieto J, et al. catheters. Pigtails catheters are preferable to straight ClinSafety Gastroenterol and efficacy Hepatol of endoscopic 2015; 13:747-52.ultrasound-guided [PMID: 25290534] drainage of pancreatic stents, because their complication rate is markedly lower. fluid collections with lumen-apposing covered self-expanding metal stents. 16. attempt to bridge via endoscopic stenting may contribute 66:2192.Zhu H, [PMID: Lin H, 28314736] Jin Z, Du Y. Re-evaluation of the role of lumen-apposing metal stents (LAMS) for pancreatic fluid collection drainage. Gut 2017; toFinally, pseudocyst in cases resolution.of associated In pancreatic summary, duct most disruption pancreatic an 17. pseudocysts should be managed principally by endoscopic et al. Clinical evaluation of a novel lumen-apposing metal stent for procedures and laparoscopic or open surgical approach endosonography-guidedItoi T, Binmoeller KF,pancreatic Shah J, pseudocyst Sofuni A, and Itokawa gallbladder F, Kurihara drainage T, (with videos). 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