Choledochoduodenal fistula: a rare complication of acute peptic ulcer bleeding

Abstract

A 75-year-old man presented with a 3-week history of melaena and right upper quadrant pain. This was on a background of significant alcohol intake and a complex medical history. He was haemodynamically unstable with investigations indicating a new iron-deficiency anaemia. After resuscitation, urgent intervention was required under general anaesthesia. This involved a triple phase abdominal CT, followed by emergency oesophagogastroduodenoscopy. This revealed deep ulceration with extension to the pancreatic head and common bile duct. There was also evidence of pneumobilia on CT, secondary to a choledochoduodenal fistula. Treatment encompassed an invasive and medical approach. Following treatment, the patient was stable, with follow-up endoscopy exhibiting good duodenal mucosal healing.

Background

Choledochoduodenal fistulas (CDFS) are a very rare complication of gastrointestinal disease, with only 5% of fistulas presenting as a consequence of duodenal ulcers.¹ However, studies have shown that these fistulas tend to be incidental findings and that there are no specific symptoms of diagnostic value.² Acknowledgement is vital as complete resolution of the ulcer is dependent on healing of the CDF.³ This case report identifies the need for early imaging to thoroughly evaluate and identify CDFs in elderly individuals with a background of gastrointestinal and biliary disease. Specifically, gastrointestinal haemorrhage and a concomitant presence of pneumobilia on CT should prompt a high index of suspicion.

Case presentation

A clinically unwell 75-year-old man presented with a 3-week history of melaena with associated right-sided chest and abdominal pain without vomiting.

This was on a background of extensive comorbidities evidencing the gentlemen’s frailty. Previous endoscopy indicated grade 1 oesophageal varices, secondary to portal hypertension due to alcohol related liver cirrhosis. Other chronic gastrointestinal conditions included ileocolonic Crohn’s with previous resection and bile acid diarrhoea. He also underwent endovascular aortic repair earlier in the year for an infrarenal abdominal aortic aneurysm.

The patient lived alone and was fully independent with his activities of daily living, with a heavy alcohol consumption of approximately 42 units per week.

On examination, he was hypotensive with a blood pressure of 88/47 mm Hg and heart rate of 110 beats per minute. A digital rectal examination showed evidence of melaena and fresh blood, necessitating urgent invasive investigations following patient stabilisation. The patient received two units of packed red blood cells, fresh frozen plasma and intravenous vitamin K therapy. Prior to endoscopic intervention, the patient was intubated and received vasopressor support periprocedure.

Investigations

Laboratory tests corroborated a new iron deficiency anaemia (haemoglobin 55 g/L, baseline 111 g/L; transferrin saturations 7%) with a raised urea (14.7 mg/dL) and acute kidney injury (creatinine 143 µmol/L, baseline 70 µmol/L). However, his liver function and clotting function was normal, with a venous lactate was 1.2. To exclude postvascular surgery complications, an urgent CT was organised. This showed pneumobilia within the common bile duct and biliary tree (figure 1A, B). There was deep ulceration to the serosal layer of the second part of the duodenum (D2) with extension to the pancreatic head and common bile duct, with a normal ampulla seen. Endoscopic evaluation showed a 25 mm deep cratered anterio-medial ulcer at the D1/D2 junction, with an adherent clot (figure 1C, D). Adjacent to this was a CDF with bile reflux into the duodenal lumen. There was no evidence of duodenal adenoma or diverticulum seen on cross-sectional imaging or endoscopy.

Figure 1.

CT showed pneumobilia (arrow) within the common bile duct and biliary tree (A) and deep ulceration to the serosal layer of D2 (arrow) with extension to the pancreatic head and common bile duct (B). On endoscopy, there was evidence of choledochoduodenal fistula (arrow) with bile reflux into the duodenal lumen adjacent to the ulcer (C). An underlying visible vessel (arrow) was treated with endoscopic clipping and epinephrine injection (D).

Treatment

The underlying visible vessel was successfully treated with endoscopic clipping as epinephrine injection and coagulation therapy failed to achieve haemostasis. Postendoscopy the patient was extubated after 4 hours of high-dependency monitoring and did not require intensive care support. After hepatopancreatobiliary multidisciplinary (MDT) review, the fistula was managed medically with continuous intravenous proton pump infusion of intravenous pantoprazole (8 mg/hour for 72 hours), empiric Helicobacter pylori eradication therapy and total parental nutrition.

Outcome and follow-up

After 7 days, he tolerated an oral diet and was discharged home, with repeat endoscopy at 6 weeks showing duodenal mucosal healing (figure 2).

Figure 2.

Endoscopic mucosal healing of duodenal ulcer at D1/D2 junction at 6 weeks from initial presentation.

Discussion

CDFs are an atypical connection between the common bile duct and duodenum. It is a very rare complication of peptic ulcer diseases as well as cholelithiasis.⁴ Gong et al have classified it based on its location with type A CDF located more than 2 cm from the duodenal papilla and type B located less than 2 cm from the papilla.⁵ The association between peptic ulcer disease and CDF is thought to be attributed to the fact that majority of duodenal ulcers are located in the first part of the duodenum, and that the common bile duct is in close proximity.⁶

CDF presents with non-descript symptoms but includes abdominal pain, vomiting and jaundice.⁷ However, they have always remained a diagnostic challenge due to its ambiguous presentations, despite advances in imaging allowing for increased cases to be reported.⁸ Shimono et al conducted a study to look at using CT to differentiate CDF from gallbladder enteric fistulae and emphysematous cholecystitis. In this study, it was concluded that though the CT scan could not detect the fistula directly, all the patients with CDF had air in the common bile duct on CT.⁷ This was true in this case report as well. The presence of pneumobilia as a diagnostic indicator for CDF has been substantiated in other reports, either on abdominal X-ray, CT or ultrasound.^{2 9–11}

There is no clear consensus on principles of management.¹¹ However, Zong et al claim that prompt management is required to prevent recurrence of the ulcer.¹² Size also seems to be key in dictating management as Li et al claimed that fistula orifices below 0.5 cm required medical management and those above required forms of surgical intervention.¹³ Other literature argues that management depends on the underlying aetiology.¹⁴ Fistulae’s caused by ulcer disease are thought to have good outcomes if treated medically with antacid and inhibitors of acid secretion. This could be because the fistulae orifice is thought to be smaller than those complicating cholelithiasis.³ Surgical intervention is usually necessary if the patient complains of recurrent ulcers or failure to control symptoms. However, as hepatico-pancreato-biliary surgery is complex and associated with higher morbidity, a medical approach may be preferred. Hence, management is dependent on a range of factors, and it is important an MDT approach is employed for holistic care. This was done in this case as owing to patient factors and the character of the CDF, a medical approach was deemed more suitable than surgical. As the patient showed signs of improvement, a surgical revision was not needed.

In conclusion, this case report highlights the clues pertinent in diagnosing a CDF and the principles of management that must be considered.

Learning points.

• A high index of suspicion for a choledochoduodenal fistula (CDF) is needed in patients with gastrointestinal haemorrhage and pneumobilia on CT.

• A medical management strategy can be very effective with regard to outcomes of CDF.

• In patients with CDF, a multidisciplinary team approach must be taken to effectively manage this condition.

Ethics statements

Patient consent for publication

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