Introduction: We present a patient with an unusual cause of biliary obstruction.
Case Report: A 50-year-old male was presented with a five-month history of worsening recurrent biliary abdominal pain and fevers. There was no previous biliary surgery. His workup revealed a normal bilirubin with elevation of other liver tests. Abdominal ultrasound demonstrated a common bile duct (CBD) diameter of 6 mm and cholelithiasis. A magnetic resonance cholangiopancreatography was unremarkable. An endoscopic ultrasound showed gallbladder sludge and stones, as well as CBD wall thickening with sludge in its mid to distal segments. At endoscopic retrograde cholangiopancreatography, a CBD filling defect was noted. After sphincterotomy, a balloon catheter extracted what looked like a cast occupying the entire lower CBD, extending into the cystic duct. This was retrieved in one piece using a rat tooth forceps and sent for pathology. The patient was discharged without complication. Cholecystectomy was recommended. Pathological analysis revealed the concretion was made of vegetable material. There have only been eight cases of biliary phytobezoar described in the modern English medical literature. Most reports describe the occurrence of a biliary phytobezoar presenting up to 40 years following a surgical bilioenteric anastomosis either with associated choledocholithiasis or alone. There exist only two case reports of patients having developed a biliary phytobezoar in the absence of any bilioenteric anastomosis or fistula. In both, the bezoar acted as a nidus for CBD stone formation, although the mechanism for developing a phytobezoar is not completely understood.
Conclusion: We describe the first reported case of an isolated biliary phytobezoar in the absence of previous biliary surgery or bilioenteric fistula.

Keywords: Endoscopic retrograde cholangiopancreatography (ERCP), Phytobezoar, Post-biliary surgery

Bezoar is defined as a foreign body resulting from accumulation of ingested material and classified according to its composition.

A phytobezoar is the most common type of bezoar, and is composed of indigestible vegetable-like material. Phytobezoars are commonly reported in patients who have had previous gastric surgery [1][2]. They can occur at any site in the gastrointestinal tract; but most commonly are found in the stomach. However, biliary phytobezoars are extremely rare. We describe, for the first time, a case of a patient presenting with an obstructing biliary phytobezoar causing cholangitis in the absence of a history of abdominal surgery, sphincterotomy, spontaneous biliary-enteric fistula, or associated choledocholithiasis.

A 50-year-old male was presented with a five-month history of worsening recurrent biliary abdominal pain and fevers. There was no significant past medical history, including no previous biliary surgery. Physical examination showed only right upper quadrant tenderness.

Workup of the patient revealed a total bilirubin of 7.4 mg/L, direct bilirubin 4.8 mg/L, aspartate aminotransferase 71 U/L, alanine aminotransferase 103 U/L, alkaline phosphatase 292 U/L, and gamma–glutamyl transferase 992 U/L. Abdominal ultrasound demonstrated a common bile duct (CBD) diameter of 6 mm, pneumobilia and cholelithiasis. A magnetic resonance cholangiopancreatography (MRCP) from another institution was reported as unremarkable. An endoscopic ultrasound (EUS) showed gallbladder sludge and stones, as well as CBD wall thickening with sludge in its mid to distal segments. At endoscopic retrograde cholangiopancreatography (ERCP), a CBD filling defect was noted. After sphincterotomy, a balloon catheter extracted what looked like a cast occupying the entire lower CBD, extending into the cystic duct (Figure 1). This was retrieved in one piece using a rat tooth forceps and sent for pathology. The patient was discharged without complication. Pathological analysis revealed the cast was made of vegetable material (Figure 2).

A literature search revealed only eight cases of biliary phytobezoar described in the modern English medical literature. Four were isolated biliary phytobezoars [2][3][4][5], while the phytobezoar acted as a nidus for CBD stones in the other patients [2][6][7][8] (Table 1).

In 1972, Ban et al. [6] described a biliary phytobezoar acting as nidus for symptomatic CBD stones that had developed in a patient two years post- choledochojejunostomy. Most reports describe the occurrence of a biliary phytobezoar presenting up to 40 years following a surgical bilioenteric anastomosis either with associated choledocholithiasis [9] , or alone [2][3][5] in one case as a result of a choledochoduodenal fistula 12 years post cholecystectomy [4]. There exist only two case reports of patients having developed a biliary phytobezoar in the absence of any bilioenteric anastomosis or fistula. In both, the bezoar acted as a nidus for CBD stone formation [7][8] as has also been noted with foreign bodies such as surgical clips [10].

Although the mechanism for developing a phytobezoar is not completely understood, a main contributing factor relates to ablation or bypass of the sphincter of Oddi due to surgical manipulation or fistula formation. The mechanism in the absence of any such altered anatomy remains unclear and some have suggested that intermittent stone passage may contribute [7]. Sphincter of Oddi manometry abnormalities were noted in one patient, and remain of unclear clinical significance [9]. In the current patient, although cholelithiasis was noted, no CBD stone was present in the phytobezoar. So, cholecystectomy was recommended.

In conclusion, we describe the first reported case of an isolated biliary phytobezoar in the absence of previous biliary surgery or bilioenteric fistula.

1. Angelelli G, Magliocca M, Zaccheo N, Vinci R, Rotondo A. Intestinal obstruction caused by phytobezoar: Computerized tomography findings. Report of 3 cases. [Article in Italian]. Radiol Med 1997 Jun;93(6):789–91.   [Pubmed]    
2. Kim Y, Park BJ, Kim MJ, et al. Biliary phytobezoar resulting in intestinal obstruction. World J Gastroenterol 2013 Jan 7;19(1):133–6.   [CrossRef]   [Pubmed]    
3. Lamotte M, Kockx M, Hautekeete M, Holvoet J, Hubens H. Biliary phytobezoar: A medical curiosity. Am J Gastroenterol 1995 Aug;90(8):1346–8.   [Pubmed]    
4. Moghaddam JA, Amini M, Adibnejad S. Development of bile duct bezoars following cholecystectomy caused by choledochoduodenal fistula formation: A case report. BMC Gastroenterol 2006 Jan 5;6:1.   [Pubmed]    
5. Bae JM, Lee YK. Extremely rare case of extrahepatic duct phytobezoar treated with intraoperative transenteral endoscopy. Ann Surg Treat Res 2014 Aug;87(2):100–3.   [CrossRef]   [Pubmed]    
6. Ban JL, Hirose FM, Benfield JR. Foreign bodies of the biliary tract: Report of two patients and a review of the literature. Ann Surg 1972 Jul;176(1):102–7.   [CrossRef]   [Pubmed]    
7. Cetta F, Lombardo F, Rossi S. Large foreign body as a nidus for a common duct stone in a patient without spontaneous biliary enteric fistula or previous abdominal surgery. HPB Surg 1993;6(3):235–42.   [Pubmed]    
8. Kim TO, Lee SH, Kim GH, et al. Common bile duct stone caused by a phytobezoar. Gastrointest Endosc 2006 Feb;63(2):324; discussion 324.   [CrossRef]   [Pubmed]    
9. Procházka V, Krausová D, Kod'ousek R, Zámecníková P. Foreign material as a cause of choledocholithiasis. Endoscopy 1999 Jun;31(5):383–5.   [CrossRef]   [Pubmed]    
10. McMahon GS, Attar S, Dennison AR. Bile duct "clip-stones": Why a stitch in time could save nine. Hepatogastroenterology 2010 Sep–Oct;57(102–103):1037–9.   [Pubmed]