An unusual cause of acute internal haemorrhage: cystic artery pseudoaneurysm secondary to acute cholecystitis

Case

A 64-year-old male was admitted under the emergency general surgical team with a 1-day history of epigastric pain and no change in bowel symptoms.

His past surgical history included laparotomy for small bowel volvulus that did not require bowel resection, laparoscopic appendicectomy, open right inguinal hernia repair and traumatic laceration to testes with subsequent bilateral orchidectomy. In 1993, he was investigated for upper abdominal pain and abdominal ultrasound that showed possible cholesterolosis. A diagnostic ERCP was normal. He was asymptomatic from his biliary pathology until a recent episode of CT-proven cholecystitis, so was placed on the waiting list for an elective laparoscopic cholecystectomy.

His medical co-morbidities included ablation of an atrio-ventricular nodal re-entry tachycardia.

On clinical examination, he was dehydrated but not jaundiced, with tenderness in the epigastrium and no peritonitis. The results of admission investigations are summarised in Table 1. He was managed as acute pancreatitis and cholecystitis with intravenous fluids, urinary catheter and intravenous antibiotics.

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Table 1.

Admission haematological, biochemical and radiological results.

Admission blood parameters
• WCC	14.7 × 109/L
• Bilirubin	23 µmol/L
• Alanine transaminase	69 units/L
• Alkaline phosphatase	211 units/L
• Gamma glutamate	207 units/L
• Amylase	1247 units/L
• C-reactive protein	63 mg/L
Admission erect chest X-ray	No free air under diaphragm

Ultrasound showed a large calculus in the neck of a thickened gallbladder and intra-hepatic duct dilatation.

Whilst awaiting an MRCP to investigate for choledocholithiasis as a cause of acute pancreatitis and cholecystitis, the patient developed coffee ground vomiting without haemodynamic compromise. His haemoglobin was stable at 10 g/dL. A nasogastric tube was inserted and placed on low-grade suction. Only 22 mL was aspirated over 24 hours. However, there was a subsequent drop in haemoglobin to 8 g/dL and an urgent CT angiogram was organised to exclude suspected splenic artery pseudo-aneurysm haemorrhage as a complication of acute pancreatitis.

CT examination revealed a thick-walled gallbladder with pseudoaneurysm of the cystic artery (Figures 1 and 2), collapsed inferior vena cava suggesting hypovolaemia and free fluid in the peritoneal cavity highly suspicious of blood (Figure 3). The patient was taken to theatre for an urgent laparotomy where there was active haemorrhage from the cystic artery with free blood in the peritoneal cavity. There was gallbladder perforation with gallstones free in the peritoneal cavity. The cystic artery was suture ligated to control the haemorrhage. A subtotal cholecystectomy was performed and a drain was placed in the sub-hepatic area. Post-operatively, the patient was monitored in the high dependency unit and the patient made an uneventful recovery. He was discharged on day 15 to await an outpatient MRCP. Histopathology revealed severe acute-on-chronic xanthogranulomatous cholecystitis with no dysplasia or malignancy. OPEN IN VIEWER

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Figure 2.

Coronal image of the abdomen and pelvis following administration of the intravenous contrast showed a round well-defined area of extravasation of contrast in an abnormal oedematous gallbladder which contained a large gallstone. The findings were those of pseudoaneurysm of cystic artery with bleeding into the inflamed gallbladder. Note the free blood around the liver and in the pelvis.

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Figure 3.

Axial post-intravenous contrast CT image of the cystic artery pseudoaneurysm. Note the collapsed IVC indicating hypovolaemia and state of shock.

Discussion

Spontaneous cystic artery haemorrhage is a rare condition.^1–9 This condition has been previously reported in the context of blunt abdominal trauma,^10–12 von Willebrand’s disease, metachromatic leukodystrophy and polyarteritis nodosa. ¹³ Additionally, cystic artery haemorrhage is known to occur in the presence of cholecystitis, possibly as a consequence of pseudoaneurysm formation. ¹⁴ The paucity of cystic artery pseudoaneurysms despite the high incidence of cholecystitis may reflect early occlusion of the artery as part of the inflammatory process.^7,15 In this case report, we describe a patient who had spontaneous cystic artery haemorrhage and non-traumatic gallbladder perforation in the presence of acute cholecystitis.

The diagnosis of cystic artery haemorrhage can be challenging. Quinke’s clinical triad of upper abdominal pain, jaundice and upper gastro-intestinal bleeding in the presence of known gallstones might suggest the diagnosis. ³ Endoscopy might show haemobilia, ¹⁴ whilst contrast CT might reveal pooling of contrast in the gallbladder lumen.^10,11,14 Endoscopic retrograde cholangiopancreaticography (ERCP) may show filling defects from blood clots within the common bile duct. ¹⁵

The diagnosis can be confirmed with intra-arterial angiography, which could also enable selective embolisation of the bleeding vessel.^8,15 In our patient, an urgent CT angiogram was performed to identify splenic artery pseudoaneurysm haemorrhage as a cause for acute gastro-intestinal bleeding. We note the alternative option in this situation could be urgent upper gastro-intestinal endoscopy to identify stress ulcers or gastritis but this would not have identified the pseudoaneurysm and would have delayed the diagnosis.

Selective angiographic embolisation of cystic artery haemorrhage has been reported with good outcomes.^8,10,14–16 Interval cholecystectomy may be performed^14,15 but is not essential.^8,10,16 In our patient, we proceeded to theatre after resuscitation. We decided not to undergo selective angiography and embolisation because the patient had clinical and radiological evidence of active cystic artery haemorrhage and we believed operative haemorrhage control was indicated. Additionally, this approach enabled concurrent cholecystectomy.

Histopathological examination of the resected gallbladder demonstrated xanthogranulomatous cholecystitis, a finding only reported twice in the scientific literature in the presence of cystic artery pseudoaneurysms.^17,18

Conclusion

Spontaneous cystic artery haemorrhage is a rare complication of acute cholecystitis.