Rupture of portal vein pseudoaneurysm caused by impinging infected walled off pancreatic necrosis (WOPN): a rare complication
- 1 Department of Surgical Gastroenterology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
- 2 Department of Gastroenterology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
- Correspondence to Professor Rajesh Gupta; rajsarakshi@gmail.com
Abstract
We report a case of walled off pancreatic necrosis in a patient with alcoholic pancreatitis who underwent endoscopic ultrasound-guided multiple pigtail catheter drainage. 10 days later patient presented with massive haemorrhage likely due to erosion of portal vein pseudoaneurysm caused by decubitus of pigtails. Patient required emergent portal venorrhaphy to arrest haemorrhage.
Background
Venous complications in acute pancreatitis are mostly related to thrombosis of splenic, portal or superior mesenteric veins.1 Venous aneurysm in setting of acute pancreatitis is extremely rare. Most cases of portal vein aneurysm (PVA) (63%) are extrahepatic and most common aetiology is congenital though acquired vascular abnormality or degenerative pathology has also been reported.2 Various causes of acquired PVA include portal hypertension with cirrhosis, trauma and invasion of the portal vein by malignancy.2 Portal vein pseudoaneurysm in setting of acute pancreatitis is anecdotal. We present a rare case of rupture of portal vein pseudoaneurysm in the setting of infected walled off pancreatic necrosis (WOPN).
Case presentation
A 35-years alcoholic man, underwent Endoscopic Ultrasound guided (EUS)-guided cystogastrostomy and necrosectomy with placement of two plastic double pigtail stents through posterior gastric wall during second week of illness and three double pigtail stents through antropyloric region during third week of illness (figure 1). Patient showed improvement with reversal of organ dysfunction and resolution of necrotic cavity and was discharged after 1 month of hospital stay. Contrast Enhanced Computerised Tomogram (CECT) at time of discharge showed largely collapsed WOPN cavity, however, an outpouching of portal vein was noted for which no treatment was offered as patient was asymptomatic (figure 2). Ten days later, patient presented with warning bleed in the form of melaena followed by massive haematemesis and hypovolaemic shock with haemoglobin of 2.9 g/L at presentation.
Walled off necrosis with portal vein wall near the confluence has an irregular outline (arrow).
Portal vein showing an outpouching of the wall (arrow) with partially drained necrotic cavity.
Differential diagnosis
Clinically we suspected the bleed from pseudoaneurysm arising from either splenic artery or gastroduodenal artery which is more common in WOPN after double pigtail stents.
Investigations
CT scan done after presentation with melaena revealed outpouching of the portal vein and no arterial anomaly and no extravasation of contrast (figure 3). Twenty-four hours later patient presented with massive haematemesis for which exploratory laparotomy was performed in view of haemodynamic instability.
Multiple transgastric plastic stents noted, arrow shows outpouchings from portal vein.
Treatment
On exploration, we found a large haematoma in lesser sac bulging through gastrocolic omentum which was evacuated along with necrosum. In view of massive bleeding and obscured view, we packed the cavity with sponges. We noticed cystogastrostomy in posterior gastric wall and antropyloric region with five double pigtail plastic stents (DPS) in situ.
After resuscitation of the patient, sponges were removed and we noted 1.0×1.0 cm rent in the anterior wall of the portal vein at splenoportal junction. The antropyloric DPS stents were retrieved from portal vein rent. We failed to control bleeding with vascular clamp as tissues in the area were inflamed, friable and oedematous. The assistant applied digital pressure over the rent in the portal vein to achieve haemostasis and interrupted 4–0 prolene sutures with half circle, 20 mm needle were applied in synchronisation with the assistant (figures 4–6). All stents lying in the field were removed and gastrostomy and antropyloric stent sites repaired with interrupted 3–0 polypropylene sutures. There was ~2.5 L of blood loss intraoperatively requiring multiple blood transfusions.
Bleeding from the portal vein which was controlled by digital pressure.
Difficulty in suture placement, addressed by digital pressure.
Vein was sutured by 4–0 prolene in synchronous manner by applying digital pressure.
Outcome and follow-up
Postoperatively patient required organ support and nutritional support in intensive care unit. Patient underwent tracheostomy in view of anticipated prolonged ventilatory support. Patient developed bile leak (300 mL/day) and abdominal dehiscence that was managed conservatively. Doppler study done postoperatively showed no flow in the portal vein, however, CT scan showed mild narrowing at splenoportal junction with opacification of main portal vein (figure 7). Patient was discharged after 60 days of hospital stay with both drains in situ which were removed in follow-up when output reduced below 10 mL/day and drain fluid amylase was normal. Patient is 1 year postoperative and doing well with no exocrine and endocrine insufficiency.
CT image showing opacification of the portal vein, arrow showing irregularity in the portal vein at the sutured site.
Discussion
Focal aneurysmal dilatation of portal vein is a rare entity. It can be categorised as congenital, acquired (portal hypertension, trauma, tumour invasion) and pseudoaneurysm (secondary to necrotising pancreatitis).1–3 There is one report in the literature of portal vein pseudoaneurysm caused by impinging pseudo cyst.4 5 Present case is the first in the literature reporting rupture of portal vein pseudoaneurysm caused by impinging WOPN.
Arterial pseudoaneurysms are more common than venous aneurysm in patients with DPS in WOPN and are much more common when lumen apposing metal stents (LAMS) were used (8.7% in LAMS vs 0.7% in DPS).6 Diagnosis of pseudoaneurysm of portal vein can be challenging as subtle irregularity in the wall of portal vein was the only sign visible on CT scan initially (figures 1 and 2). However, after EUS-guided drainage of WOPN, subsequent CT scan revealed outpouching of weakened portal vein (figure 3), which probably was due to sudden decrease in the intracystic pressure by DPS stents. Patient subsequently presented with warning bleed in the form of melaena and 24 hours later with massive haematemesis. Though it is difficult to pinpoint the cause of rupture but it is likely due to residual necrosum or decubitus of transgastric pigtail stents.
Management of PVA depends on aetiology and presence or absence of complications. Congenital PVA are likely to have uncomplicated course compared with acquired PVA. Uncomplicated PVA has been treated conservatively previously.2 However, surgery is warranted for PVA presenting with bothersome symptoms, increase in size over time, mass effect, rupture or for non-thrombotic PVA >3 cm.7 8 On the other hand pseudoaneurysms of portal vein would merit intervention due to high risk of rupture.
There are two cases of portal vein erosion in acute pancreatitis described in literature, one case was managed with exploratory laparotomy and initial packing of abdominal cavity for haemostasis and subsequently percutaneous stent graft placement. Other case was managed conservatively.9 However, portal vein pseudoaneurysm occurring in the setting of WOPN managed with DPS stent, cannot be left alone. It is important to highlight that our patient recovered for 3 weeks and then presented with rupture.
Surgery to control bleeding from portal vein in necrotic cavity has its own challenges due to presence of friable, inflamed and oedematous tissues which are likely to give in to the compression of vascular clamps and may further damage the vein. We realised that in such a scenario best way to control haemorrhage was to apply digital pressure on the rent itself. Digital compression of portal vein above and below the rent was ineffectual as bleeding from splenic vein side was continuous and obscured the narrow operative field. One has to suture in a synchronised way as assistant lifts digital pressure from the rent and surgeon takes sutures and immediately assistant puts back digital pressure to occlude the rent thus limiting blood loss. We found that half circle, 20 mm size, needle with 4–0 polypropylene was helpful as this size needle was easy to visualise and handle in presence of bleeding. One has to be careful while taking suture and tying knots as suture can cut through inflamed wall of portal vein. At the same time, care has to be taken not to accidently take posterior wall. It will be prudent to remove any residual necrosum in the cavity. This case also brings to attention the fact that presence of portal vein pseudoaneurysm in WOPN with DPS stents in vicinity should be subjected to definitive management (endovascular/surgical) depending on availability of expertise even if patient is asymptomatic.
To summarise, haemorrhage following endoscopic interventions in acute necrotising pancreatitis occurs most commonly from rupture of arterial pseudoaneurysm, however, in the present case we found that haemorrhage occurred from rupture of portal vein pseudoaneurysm following endoscopic transgastric pigtail catheter drainage of WOPN. The pseudoaneurysm was picked up on CT scan and patient underwent emergent laparotomy and venorrhaphy to control exsanguinating haemorrhage.
Patient’s perspective
Acute pancreatitis is a deadly disease, I went through multiple blood transfusions, required tracheostomy and frequent suctioning of secretions from tracheostomy which I got rid after 3 to 4 months. At the end, I am alive now and able to do routine activities after 5 months of surgery. I really thank my doctors and family for their care and support.
Learning points
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Infected walled off pancreatic necrosis impinging on portal vein can lead to pseudoaneurysm which is liable to erosion by decubitus of a pigtail and residual necrosum.
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Management is required even in asymptomatic patients due to risk of rupture and bleed.
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Management of rupture is emergent laparotomy, pressure haemostasis, suture venorrhaphy, removal of residual necrosum and transgastric pigtails and lesser sac drainage with soft silicone drains.
Footnotes
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Contributors Once the patient was recovered post surgery RG and VBM have decided to report this case. VBM collected the data and manuscript was contributed by VK and SR. Final changes in the manuscript and images were done by RG and VBM.
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Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
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Competing interests None declared.
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Patient consent for publication Obtained.
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Provenance and peer review Not commissioned; externally peer reviewed.
- © BMJ Publishing Group Limited 2021. No commercial re-use. See rights and permissions. Published by BMJ.
References
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