Case Reports in Gastrointestinal Medicine

Case Reports in Gastrointestinal Medicine / 2021 / Article

Case Report | Open Access

Volume 2021 |Article ID 6610610 | https://doi.org/10.1155/2021/6610610

Hussam I. A. Alzeerelhouseini, Muawiyah Elqadi, Mohammad N. Elqadi, Sadi A. Abukhalaf, Hazem A. Ashhab, "Endoscopic Drainage of Giant Pancreatic Pseudocysts Using Both Lumen-Apposing Metal Stent and Plastic Stent: A Report of Two Cases and Review of the Current Literature", Case Reports in Gastrointestinal Medicine, vol. 2021, Article ID 6610610, 12 pages, 2021. https://doi.org/10.1155/2021/6610610

Endoscopic Drainage of Giant Pancreatic Pseudocysts Using Both Lumen-Apposing Metal Stent and Plastic Stent: A Report of Two Cases and Review of the Current Literature

Academic Editor: Yoshifumi Nakayama
Received14 Oct 2020
Revised22 Jan 2021
Accepted16 Mar 2021
Published02 Apr 2021

Abstract

Introduction. A pancreatic pseudocyst (PP) with major diameter equal to 10 cm or more is called a giant pseudocyst. The ideal management for giant PPs is controversial. Endoscopic drainage is an alternative nonsurgical approach for PP management. Only a few cases of giant PPs were reported to be managed by endoscopic drainage. Case Presentation. We reported two cases of giant PPs following an episode of acute pancreatitis. Both were resolved following endoscopic cystogastrostomy using metallic and double-pigtail stents with excellent outcomes. There was no history of recurrence or complications on follow-up. In addition, we extensively reviewed all available literature studies of giant pancreatic pseudocyst presentation, management, and complications. We summarized all reported cases and presented them in a comprehensive table. Conclusion. The endoscopic cystogastrostomy approach is cost saving, can avoid surgical complications, and offers an early hospital discharge.

1. Introduction

Pancreatic pseudocyst (PP) is a peripancreatic fluid collection (PFC) and is a well-known complication of pancreatic diseases. PP usually occurs four weeks after acute pancreatitis and is a rich amylase fluid collection surrounded by a well-defined wall lacking solid material [1]. Worldwide, PP cases are increasing lately due to an increase in acute pancreatitis hospitalization over time [2] and advanced radiological modalities [3].

The management decision of PPs relies on clinical and imaging evaluation. A majority of PPs are asymptomatic with observation till spontaneous regression is required. However, symptomatic, persistent, large, or complicated pseudocysts require internal drainage [4, 5]. A pseudocyst with a major diameter equal to 10 cm or more is called a giant pseudocyst [6].

This intervention can be performed surgically or using less-invasive percutaneous or endoscopic approaches [7]. Endoscopic drainage of PPs is an alternative nonsurgical approach. Since the first reports by Sahel et al. [8] and Cremer et al. [9], endoscopic drainage of PPs has become an established procedure; it entails the creation of a fistulous tract between the PP and the gastric lumen (cystogastrostomy) or duodenal lumen (cystoduodenostomy) [8, 9].

Herein, we describe two cases in which giant pancreatic pseudocysts were resolved following endoscopic cystogastrostomy. Only a few cases of giant pancreatic pseudocysts were found in the literature review.

2. Case Presentation

2.1. Case 1

A 63-year-old female was admitted to our hospital complaining of abdominal pain and vomiting. She was diagnosed as a case of acute idiopathic pancreatitis 4 weeks before her admission which was treated conservatively. On general examination, she was not icteric or feverish. Abdominal examination revealed a palpable, firm, tender epigastric mass and otherwise unremarkable. Laboratory investigations revealed markedly elevated serum amylase with normal lipase. White blood cell (WBC) count and liver enzymes were within normal limits. Chest X-ray (CXR) showed left-side moderate pleural effusion. An abdominal CT scan showed a huge pancreatic pseudocyst measuring 15 cm  15 cm  12 cm, occupying the body and tail of the pancreas (Figure 1(a)).

Endoscopic ultrasound- (EUS-) guided drainage was performed as a therapeutic procedure using a Pentax linear echoendoscope. On the EUS, there was a huge fluid collection with air and debris consistent with the infected pancreatic pseudocyst (Figure 2(a)). A 10 mm length, 15 mm diameter Hot AXIOS stent was placed with EUS guidance with immediate drainage of >2000 cc of fluid and debris (Figures 2(b) and 2(c) and Figure 3). Then, a 10 French gauge, 1 cm long double-pigtail stent was placed under fluoroscopy guidance. The patient tolerated the procedure well, and there were no complications. One day after the procedure, a CT scan showed excellent results with regression of the pseudocyst. The patient was discharged home on antibiotics (Figure 1(b)).

On 6 weeks of follow-up, the patient reported disappearance of symptoms. EUS was repeated, and both AXIOS and plastic stents were removed (Figure 4); the patient made an uneventful recovery and was discharged home on antibiotics.

2.2. Case 2

A 37-year-old female was admitted to our hospital complaining of epigastric pain radiating to the back, vomiting, and early satiety. She had a history of acute biliary pancreatitis 4 weeks prior to her admission which responded well to medical management. On examination, there was marked epigastric distention and tenderness with decreased air entry at the base of the left lung. The examination of the other systems was normal. Laboratory tests revealed high serum lipase with normal serum amylase. Complete blood count (CBC), liver enzymes, and serum electrolytes were within normal limits.

A CXR revealed mild pleural effusion on the left side. An abdominal CT scan with contrast showed a giant multiseptated cyst measuring about 20  15  8 cm in the body and tail of the pancreas consistent with the pancreatic pseudocyst (Figure 5). The head of the pancreas was enlarged and surrounded by significant fat stranding. There was a moderate amount of free fluid noted at the Morison pouch and pelvic cavity. The liver, the spleen, and both kidneys were normal in size and density.

Endoscopic ultrasound- (EUS-) guided drainage was performed to relieve this giant symptomatic pseudocyst. Periprocedural period was covered with an antibiotic. EUS showed a huge extrinsic bulge at the body and antrum part of the stomach. The Hot AXIOS stent with 10 mm  15 mm diameter was placed with excellent drainage of >1500 cc of fluid, and then a 10 French gauge, 3 cm long double-pigtail stent was placed under fluoroscopy guidance (Figures 6 and 7). The follow-up CT scan demonstrated almost total regression of the pseudocyst (Figure 8). The patient made an uneventful recovery and was discharged home on the same day on antibiotics.

On 6 weeks of follow-up, the patient was doing well. Repeated EUS showed full resolution of the pseudocyst, and both stents were removed without complications.

3. Discussion

Pancreatic fluid collections (PFCs), according to the revision of the Atlanta classification of acute pancreatitis, are described in four subtypes: acute peripancreatic fluid collection, acute necrotic collection, pancreatic pseudocyst (PPs), and walled-off necrosis (WON) [1]. Pancreatic pseudocysts are collections of high concentrations of digestive enzymes in the retroperitoneum or the peripancreatic tissue planes that are lined by fibrous tissues and may contain necrotic debris or blood but lack a true epithelial lining [2]. Pancreatic pseudocysts usually appear 4 weeks after an episode of chronic pancreatitis, acute pancreatitis, or disruption in the pancreatic duct such as blunt, penetrating trauma, or injury during surgeries [3, 10, 11] (Table 1). PPs’ incidence is relatively low [29], and they are more in chronic pancreatitis compared to acute pancreatitis [30]. PPs present usually singular in the head of the pancreas though multiple PPs may present as well [11].


Study referenceAgeSexPresentationCause of the pseudocystPleural effusionTime after pancreatitisWBCAmylaseLipaseCEA and CA19-9Cyst size on CT or US in cm (largest dimension)DiagnosisManagementComplicationsSecond drainage neededDischarge timeType of stent usedTime of stent removal after EUSNecrosectomy needed

[12]33FAbdominal pain
Abdominal distention
Anorexia
Acute pancreatitisNormalElevatedNormal10.3  9.6  9.3Abdominal computed tomography (CT)Open retrogastric cystogastrostomyNoDay 7
[13]65MAbdominal pain
Abdominal distention
Anorexia
Weight loss
Acute pancreatitisElevatedElevatedNormal25.7  15.3  10.9Abdominal computed tomography (CT)Open retrogastric cystogastrostomyYes (bleeding in day 4 POD)Day 6
[14]16MAbdominal pain
Abdominal distention
Dyspnea
Yes (bilateral)Elevated35  15  14Diagnostic laparoscopyLaparotomy (excision of the pseudocyst and Roux-en-Y pancreaticojejunostomy)No
[15]56MAbdominal pain
Abdominal distension
Abdominal mass
Acute pancreatitisYes (right)6 weeks25  17Abdominal computed tomography (CT)Open cystogastrostomyNo10 pod
[16]63FAbdominal distentionAcute pancreatitis6 weeks12Abdominal computed tomography (CT)Open cystogastrostomyNo8 pod
[17]39MAbdominal painAcute pancreatitisElevatedElevated10  8Abdominal computed tomography (CT)Laparoscopic cystogastrostomyNo21 pod
[18]37FAbdominal pain
Abdominal mass
Acute pancreatitis12 weeksNormal23  15  12Abdominal computed tomography (CT)Laparoscopic transgastric cystogastrostomyYes (pseudocyst infection)9 pod
[19]18MAbdominal massAbdominal trauma22  18Abdominal computed tomography (CT)Laparoscopic transgastric cystogastrostomyNo12 pod
[20]49MAcute pancreatitisElevatedNormal10  4Abdominal computed tomography (CT)Laparoscopic cystogastrostomyNo
[21]48MAbdominal pain
Abdominal mass
Anorexia
Early satiety
Weight loss
Chronic pancreatitisNormalElevatedElevatedNormal21Abdominal computed tomography (CT)CT-guided percutaneous drainageNo
[22]61MAcute pancreatitisElevatedElevated13Abdominal computed tomography (CT)EUS-guided drainageYes (recurrence)YesMetallic stent8 weeksYes
[22]65MAbdominal painChronic pancreatitisElevated10  8.2  4.6Abdominal computed tomography (CT)EUS-guided drainageNoNo6 daysMetallic stent6 weeksNo
[22]61FAbdominal painAcute pancreatitisElevated14Abdominal computed tomography (CT)EUS-guided drainageYes (recurrence)Yes10 days1-metallic stent
2-plastic double-pigtail stent
No
[22]53MAbdominal pain
Vomiting
Acute pancreatitis7 weeksElevatedElevated10  12  8.7Abdominal computed tomography (CT)EUS-guided drainageNoNo6 daysMetallic stent2 weeksNo
[23]50MAbdominal pain
Abdominal mass
Vomiting
Acute pancreatitisYes (right)20 weeksNormalElevated12.7  10.8Abdominal US
Abdominal computed tomography (CT)
EUS-guided drainageNoNo7 daysNo
[24]37MAbdominal painAcute pancreatitis4 weeksNormalElevatedNormal10  7Abdominal USEUS-guided drainageNoNoSame dayPlastic double-pigtail stentNo
[24]54FAbdominal pain
Fever
Acute pancreatitisNormalElevatedElevatedNormal12  7  8Abdominal computed tomography (CT)EUS-guided drainageNoNoPlastic double pigtail stent3 weeksNo
[25]54FAbdominal painAcute pancreatitis3 weeksNormal13  14Abdominal computed tomography (CT)EUS-guided drainageYes (stent migration into pseudocyst)NoSame dayLumen-apposing metal stent5 weeksNo
[26]81MAbdominal painAcute pancreatitisNo4 weeksElevated17  11Abdominal computed tomography (CT)EUS-guided drainageYes (recurrence)YesPlastic double-pigtail stent8 weeksNo
[27]74MAbdominal pain
Fever
Chronic pancreatitisYes (bilateral)10Abdominal computed tomography (CT)EUS-guided drainageNoNoSame dayPlastic double-pigtails stentNo
[3]27MAbdominal pain
Abdominal distention
Weight loss
Anorexia
Acute pancreatitisYes (right)8 weeksNormalElevated30  15  14Abdominal computed tomography (CT)EUS-guided drainageYes (recurrence)Yes (after 1 week)Same day1-lumen-apposing metal stent
2-plastic double-pigtail stent
3 weeksNo
[28]69MAbdominal pain
Abdominal mass
Chronic pancreatitis1 yearNormalNormalElevated18Abdominal computed tomography (CT)EUS-guided drainageYes (pseudocyst infection, stent migration, and recurrence)YesSame dayPlastic double-pigtail stent8 weeksYes
This study63FAbdominal pain
Abdominal mass
Vomiting
Acute pancreatitisYes (left)3 weeksNormalElevatedNormalNormal15  15  12Abdominal computed tomography (CT)EUS-guided drainageNoNoSame day1-lumen-apposing metal stent
2-plastic double-pigtail stent
9 weeksYes
This study37FAbdominal pain
Abdominal distension
Vomiting
Early satiety
Acute pancreatitisYes (left)4 weeksNormalNormalElevatedNormal20  15  8Abdominal computed tomography (CT)EUS-guided drainageNoNoSame day1-lumen-apposing metal stent
2-plastic double-pigtail stent
6 weeksNo

Most pseudocysts are small, asymptomatic, and diagnosed incidentally. However, pseudocysts can produce a wide range of clinical manifestations that can be usually ascribed to the local mass effect of the pseudocyst or the associated inflammatory response. Depending on location and size, clinical symptoms may include abdominal pain, early satiety, weight loss, persistent fevers, infection of the pseudocyst, duodenal or biliary obstruction, vascular occlusion, free rupture of the pseudocyst into the peritoneal cavity, fistula formation, or digestion of an adjacent vessel resulting in pseudoaneurysms [10]. Our study on giant pseudocysts reported abdominal pain in 91% of patients, abdominal distention in 32%, abdominal mass in 22.7%, anorexia, early satiety in 18%, weight loss in 13.6%, and vomiting and fever in 9% (Table 2).


VariableValue

AgeMean: 50.4 years, range: 16–81
Male : female ratio1 : 0.5
Cause
 Acute pancreatitis18/23 (78%)
 Chronic pancreatitis4/23 (17.4%)
 Trauma1/23 (4.3%)
Clinical presentation
 Time of pseudocyst presentation after acute pancreatitisMean: 7 weeks, range: 3–20 weeks
 Abdominal pain20/22 (91%)
 Abdominal distention7/22 (32%)
 Abdominal mass5/22 (22.7%)
 Anorexia4/22 (18%)
 Early satiety4/22 (18%)
 Weight loss3/22 (13.6%)
 Vomiting2/22 (9%)
 Fever2/22 (9%)
Laboratory investigations
 Elevated WBC4/15 (26%)
 Elevated amylase or lipase16/16 (100%)
 Normal CEA and CA19-98/8 (100%)
Diagnosis
 Abdominal CT scan22/24 (92%)
 Abdominal US1/24 (4%)
 Diagnostic laparoscopy1/24 (4%)
 Cyst size on CT or US (largest dimension)Mean: 16.7 cm (10–35 cm)
Management
(1) EUS-guided drainage (14 cases)
Time for stent removal after EUSMean: 5.8 weeks, range: 2–9 weeks
Types of stents used
 Metallic4/13
 Plastic (double pigtail)5/13
 Both4/13
Complications
 Recurrence of the pseudocyst5/14 need second drainage (35%), and only 1 case needs third drainage (7%)
 Pseudocyst infection1/14 (7%)
 Stent migration2/14 (14%)
 Necrosectomy needed3/14 (21%)
Time of dischargeMean: 2.6 days
(2) Open surgery or laparoscopic (9 cases)
Time of dischargeMean: 10 days

CEA: carcinoembryonic antigen; EUS: endoscopic ultrasound.

The diagnosis of pseudocysts is increased as radiologic methods have developed [3]. The diagnosis is usually made by CT scan, magnetic resonance imaging (MRI), or transabdominal ultrasound (US). PPs appear as intra- or peripancreatic encapsulated fluid collections [11]. Findings on the CT scan of the pancreatic pseudocyst according to the revision of the Atlanta classification of acute pancreatitis include a well-circumscribed fluid collection that is usually round or oval, the fluid collection is typically extrapancreatic, homogenous fluid density, no nonliquid components within the fluid, and a well-defined wall that completely encapsulates the fluid collection [1]. If the diagnosis is in doubt, it is crucial to differentiate between the pseudocyst and a cystic pancreatic neoplasm; sampling of the cyst fluid may be necessary [31].

To date, there is no consensus on the ideal management of pancreatic pseudocysts. Although there are no crystal clear guidelines, pancreatic pseudocysts were traditionally managed with open surgical approaches including cyst-gastrostomy, cyst-duodenostomy, Roux-en-Y cyst-jejunostomy, and external drainage [10]. Recently, due to the advancement in the endoscopic era, endoscopic drainage of pancreatic pseudocysts has been performed with variable success rates though comparable studies to the surgical approaches have not been launched, and thus, management certainty cannot be achieved [10]. A 10-patient study has demonstrated that there are no significant differences between endoscopic drainage of pancreatic pseudocysts and surgical approaches in terms of treatment success, procedural complications, or reintervention. The significant differences observed were in-hospital length of stay and the mean cost savings [32]. Another study had reported a higher complication rate in the endoscopic drainage of the pancreatic pseudocyst group [33].

One of the largest related studies had demonstrated that endoscopic drainage may be performed in acute or chronic pancreatic pseudocysts and pancreatic necrosis though with different rates of successful drainage, complications, and recurrence. The study reported that chronic pancreatic pseudocysts were found to be resolved higher than acute pancreatic pseudocysts or pancreatic necrosis groups and as a marker for a successful drainage. The authors found that pancreatic necrosis was a marker for lower success rates and higher rates of complications and recurrence [5].

Endoscopic drainage of pancreatic pseudocysts has been performed using both transpapillary and transenteric approaches. To allow complete resolution, the transpapillary approach requires that the pseudocyst communicates with the main pancreatic duct and has few septations to permit complete drainage [10]. The transenteric approach can be achieved using a gastric or duodenal wall after confirming the adherence between the selected wall and the cyst using either endoluminal bulge or endoscopic ultrasound [10]. The cyst adherence is strictly associated with cyst size; larger and huge cysts may be accessed more easily though may need a longer time to complete resolution.

The pancreatic pseudocyst that measured >10 cm in major diameter was defined as a giant pancreatic pseudocyst [6]. There are several reported giant pancreatic pseudocyst cases in the literature (Table 1), with only a few cases reporting management [3, 12, 13, 15, 18, 34]. Surgical cystogastrostomy was the most common used approach; Wang and Misra [13] and Report [15] described the surgical midline incision approach to drain giant pancreatic pseudocysts measured more than 25 cm in the major diameter; it drained 3 L and 4.5 L, respectively, and both patients recovered uneventfully. The other used less invasive methods for huge pancreatic pseudocysts included percutaneous or endoscopic cystogastrostomy and percutaneous CT-guided cystogastrostomy. Alhassan et al. [34] reported percutaneous CT-guided cystogastrostomy with good outcomes and no complications with one-year follow-up. Regarding endoscopic cystogastrostomy, it may not be appropriate for the giant pseudocyst as an initial approach in the absence of well-trained hands, and using other options may be safer [3].

Over the last decade, EUS-guided drainage has been conventionally performed for the peripancreatic fluid collection (PFC) with a plastic stent such as double-pigtail stent (DPS) and a fully covered self-expanding metal stent. However, more recently, a dedicated device, a lumen-apposing metal stent (LAMS), has been developed as an alternative for PFC [35]. LAMS has a larger-diameter lumen (10 or 15 or newer 20 mm) compared with DPSs, and this can allow more efficient drainage with less risk of stent occlusion and superimposed infection. Moreover, LAMSs are designed with a biflanged shape that allows for tissue apposition and minimizes the risk of stent migration. Also, it can provide the channel for endoscopic necrosectomy and the need for repeated endoscopies [36]. There is a reduced risk of leakage with the LAMS compared to plastic stents mainly due to dual flange anchors and the fully covered nature of the LAMS [37]. Risk of perforations is more common in the DPS than in the LAMS, which some providers attributed to the fact that dual DPSs are technically more difficult to place than LAMSs [38].

The clinical and technical success rates of LAMSs have been reported to be 93–100% and 89–100%, respectively [36], with complication rates around 10%–15% [39]. LAMSs have become the stent of choice for endoscopic drainage of PFCs by many gastroenterologists because of their easy deployment, less procedure time, and direct debridement access [36]. Several studies demonstrated that LAMSs have better efficacy and safety over plastic stents for PFC [40].

The main drawback of the LAMS is that it was found to be associated with a higher bleeding rate when compared to the DPS, including late bleeding at 3–5 weeks in the treatment of postpancreatitis pancreatic fluid collections [41]. EUS guidance with the use of color Doppler may reduce the risk of intraprocedural bleeding but would not affect delayed bleeding [42]. One hypothesis is that the LAMS remained in place even after the collapse of the collection without any movement, and this causes friction to the surrounding vasculature around the necrotic cavity promoting pseudoaneurysm formation and subsequent bleeding [41]. Another explanation is the easier access of low-pH fluid with gastric acid into the cyst cavity due to the wider lumen of the LAMS, thereby causing irritation of exposed intracavitary vessels and an increased tendency for bleeding [43]. This prompted gastroenterologists to change their practice of repeating imaging at 3 weeks to assess the cavity resolution instead of 6 weeks, followed by stent removal if the fluid collection is resolved [37].

Current literature review revealed that the LAMS with coaxially placed DPS for PP drainage has been reported to be associated with a lower risk of bleeding [39] and low rates of infection compared to the placement of the LAMS alone (0% vs. 17%) [44]. A possible explanation for these lower risks is mainly due to anchoring LAMS with coaxial plastic stent which can make the passage of solid particles and low gastric pH more difficult while continuing to allow for drainage of liquid secretions around the pigtail stent [37].

4. Conclusion

The ideal management for giant pancreatic pseudocysts is controversial. Only a few cases of giant pancreatic pseudocysts were reported to be managed by endoscopic drainage. Endoscopic drainage is an alternative nonsurgical approach for pancreatic pseudocysts’ management that lately gained a wide range of acceptance due to its lower morbidity, mortality, and costs.

Most studies showed the superiority of the lumen-apposing metal stent on the double-pigtail stent in the management of peripancreatic fluid collection. The literature showed that the addition of a coaxial double-pigtail stent to the lumen-apposing metal stent was associated with a lower rate of adverse events in endoscopic drainage of peripancreatic fluid collection.

Data Availability

The data used to support the findings of this study are available from the corresponding author upon reasonable request.

Consent was obtained from the patients, and the patients accepted the final edition of the article.

Conflicts of Interest

The authors declare that they have no conflicts of interest.

Authors’ Contributions

Hazem A. Ashhab and Hussam I. A. Alzeerelhouseini conceptualized and designed the study. Hussam I. A. Alzeerelhouseini collected the data and wrote the literature. Hussam I. A. Alzeerelhouseini, Muawiyah Elqadi, Mohammad N. Elqadi, and Sadi A. Abukhalaf wrote the paper. Hussam I. A. Alzeerelhouseini and Sadi A. Abukhalaf edited and proofread the article.

Acknowledgments

The authors are very thankful for the patient and the doctors and nurses team who worked in the gastroenterology department at Al-Ahli Hospital.

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Copyright © 2021 Hussam I. A. Alzeerelhouseini et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


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