Massive upper gastrointestinal bleeding: a rare complication of cholecystoduodenal fistula

Abstract

A biliary fistula which may occur spontaneously or after surgery, is an abnormal communication from the biliary system to an organ, cavity or free surface. Spontaneous biliary-enteric fistula is a rare complication of gallbladder pathology, with over 90% of them secondary to cholelithiasis. Approximately 6% are due to perforating peptic ulcers. Symptoms of biliary-enteric fistula varies widely and usually non-specific, mimicking any chronic biliary disease. Cholecystoduodenal fistula causing severe upper gastrointestinal (UGI) bleed is very rare. Bleeding cholecystoduodenal fistula commonly requires surgical resection of the fistula and repair of the duodenal perforation. We describe the case of a previously healthy older patient who initially presented with symptoms suggestive of UGI bleeding. Bleeding could not be controlled endoscopically. When a laparotomy was performed, a cholecystoduodenal fistula was discovered and bleeding was noted to originate from the superficial branch of cystic artery.

Background

Cholecystoduodenal fistula as one of the causes of upper gastrointestinal (UGI) bleeding is very rare. To the best of our knowledge, only four similar case reports have been published to date.

Case presentation

A 70-year-old male patient with no known comorbidities presented with complains of progressive generalised abdominal discomfort, bloating and poor oral intake. The patient was not on any known medications. Patient presented to a nearby health facility for further evaluation of his symptoms and an abdominal ultrasound was done. Apart from an ectatic suprarenal abdominal aorta (2.8 cm), it was unremarkable. There was no biliary dilatation seen, no gallbladder wall thickening nor gallstones were visualised. His liver function tests were normal. The patient’s abdominal discomfort progressed to frank epigastric pain for which he was seen at the emergency department. He was treated as acute dyspepsia and given oral proton pump inhibitors, with an early date for a surgical outpatient clinic appointment.

The patient was admitted the following day for evaluation of multiple melenic bowel movements. It was associated with lethargy as well. On examination, he was pale and hypotensive. His abdominal examination was unremarkable and digital rectal examination revealed a large amount of melena. Initial investigations revealed a drop of haemoglobin from 111 to 78 g/L. After initial resuscitation with blood products, he was started on an intravenous infusion of proton pump inhibitor. An early (<24 hours) oesophagogastroduodenoscopy (OGDS) was performed which showed a large Forrest 2C pyloric ulcer with blood clots. There was no duodenal ulcer visualised. As there were no bleeding points, haemostatic powder spray was applied.

The patient did not improve clinically with evidence of persistent UGI bleeding. A repeat OGDS was done which showed another Forrest 2A ulcer at the first portion of the duodenum (figure 1). Combination therapy using through-the-scope clip (TTSC) and haemostatic powder spray was employed (figure 2). A total of six pints of pack cells were transfused up to that point. The patient became hypotensive, tachycardic and was still showing signs of an ongoing bleed. As urgent interventional radiology facility was not available, he was sent to the operating room immediately for a laparotomy. Intraoperatively, a thickened first portion of duodenum was visualised with a cholecystoduodenal fistula noted between the body of the gallbladder and the anterior wall of the first portion of the duodenum (figure 3). The lumen of the cholecystoduodenal fistula measures 1 cm in diameter (figure 4). A Billroth type II gastrectomy and cholecystectomy was performed. On release of the gallbladder from the adhered duodenal wall, active bleeding was seen from the gallbladder mucosa (likely from the superficial branch of cystic artery), adjacent to the fistula. The previous TTSC was seen attached on the gallbladder mucosa. There were no gallbladder stones and no regional enlarged lymph nodes.

Figure 1.

Oesophagogastroduodenoscopy image showing a large Forrest 2A ulcer at the anterior wall of the first part of duodenum.

Figure 2.

Oesophagogastroduodenoscopy image showing successful through-the-scope clip placement and haemostasis.

Figure 3.

Intraoperative image. The cholecystoduodenal fistula is visualised in this image (arrow). A 2 x 1 cm perforation at the anterior wall of the first part of the duodenum is seen, as well as the through-the-scope clip on the gallbladder mucosa.

Figure 4.

Resected specimen. The specimen on the left is the gallbladder while the one on the right is the partial gastrectomy specimen with a penetrating ulcer at the anterior wall of the first part of the duodenum (arrow).

Outcome and follow-up

The postoperative period was uneventful, and the patient made a steady recovery and was discharged a few days later. The histopathological report confirmed the picture of a cholecystoduodenal fistula with perforated duodenal ulcer and evidence of chronic cholecystitis. There were no dysplastic or malignant cells seen. During his last follow-up at the outpatient surgery clinic, patient was noted to be doing well with no abdominal symptoms.

Discussion

A biliary fistula which may occur spontaneously or after surgery, is an abnormal communication from the biliary system to an organ, cavity or free surface.¹ These may be external (biliary cutaneous) or internal (bilioenteric, biliobiliary or bronchobiliary).¹ Spontaneous biliary-enteric fistula is a rare complication of gallbladder pathology, with over 90% of them secondary to cholelithiasis.² Approximately 6% are due to perforating peptic ulcers.² Other causes include malignancy of the gallbladder and biliary tract, biliary abscess and echinococcus cysts.² The most common type of biliary-enteric fistula is cholecystoduodenal fistula (61%–77%), followed by cholecystocolonic fistula (14%–17%), and cholecystogastric (6%).³ Spontaneous internal biliary fistula is found three to five times more common in women, and usually in patients above 60 years old.²

The aetiology of biliary-enteric fistula still not well defined.³ It is thought that acute inflammatory process of the gallbladder cause adhesions of the gallbladder to adjacent organs, usually the duodenum. Recurrent inflammation will cause destruction of the gallbladder and eventual erosion into adjacent organs and ultimately, a fistula formation.³ Symptoms of biliary-enteric fistula varies widely and usually non-specific, mimicking any chronic biliary disease.³ Patients usually have long standing right hypochondrium pain and/or epigastric pain.¹ Other symptoms include recurrent episodes of cholangitis with accompanying fever and/or jaundice, acute or chronic pancreatitis, gallstone ileus, flatulent dyspepsia, weight loss and rarely, gastrointestinal bleeding.³ Most internal biliary-enteric fistulas are detected incidentally during endoscopic retrograde cholangiopancreatography (ERCP) or other radiological modalities employed for investigation of biliary or pancreatic diseases.¹

The diagnosis of spontaneous internal biliary fistula can be made by employing ERCP, CT or ultrasonography of the abdomen.^{1 3} ERCP can detect biliary fistula by directly observing contrast media leak from the gallbladder cavity into adjacent organs.⁴ The cystic duct needs to be patent in order for ERCP to diagnose biliary fistula.⁴ Even though ultrasonography and CT examination might not always be diagnostic, the findings of a contracted gallbladder, pneumobilia or biliary stones may suggest the presence of internal biliary fistula.³ In our case, ultrasonography of the abdomen failed to pick up any abnormalities of the biliary system. This is likely due to the aetiology being a perforated peptic ulcer.

Bleeding from biliary tract is usually into the duodenum and patients may present with haematemesis and or melena.³ In cases of UGI bleeding with inconclusive findings on OGDS, a differential of bleeding marginal ulcer from a cholecystoduodenal fistula should be considered.³ In a haemodynamically stable patient, a CT is warranted to establish diagnosis. Cholecystoduodenal fistula causing severe UGI bleed is very rare.³ Most case reports note that the source of bleeding is usually the erosion of cystic artery, either by duodenal ulcer or gallstones.^3–5 In our patient, it is likely that his duodenal ulcer eroded into the superficial branch of cystic artery. Bleeding cholecystoduodenal fistula commonly requires surgical resection of the fistula and repair of the duodenal perforation.⁴ This is due to the significant bleeding from cystic artery which is unlikely to resolve by conservative methods. As shown in our case, even with TTSC, patient continued to have active bleeding.

In conclusion, most symptoms of internal biliary fistulas are non-specific and presentation may vary widely, making diagnosis difficult.^{1 3} It should be a differential diagnosis in patients presenting with chronic upper abdominal pain, recurrent episodes of cholangitis, gallstone ileus or even gastrointestinal bleeding. A high index of suspicion coupled with the appropriate imaging modality might help in establishing a diagnosis preoperatively. In a stable patient, an ERCP is likely the investigation of choice, owing to its high sensitivity and specificity.^{1 4} In an unstable patient, prompt operative intervention is crucial.

Learning points.

• Internal biliary fistula should be a differential diagnosis in patients presenting with chronic upper abdominal pain, recurrent episodes of cholangitis, gallstone ileus or even gastrointestinal bleeding.

• A high index of suspicion coupled with the appropriate imaging modality might help in establishing a diagnosis preoperatively.

• In a stable patient, an endoscopic retrograde cholangiopancreatography is likely the investigation of choice, owing to its high sensitivity and specificity.

• In an unstable patient, prompt operative intervention is crucial.

• Bleeding cholecystoduodenal fistula commonly requires surgical resection of the fistula and repair of the duodenal perforation.