Benign biliary papillomatosis in a patient with a choledochal cyst presenting as haemobilia: a case report

Abstract

Biliary papillomatosis is a rare condition usually detected on imaging or postoperative histopathology. It may be asymptomatic or present with features of cholangitis. We report the management of a patient presenting with haemobilia.We report a rare case of benign biliary papillomatosis (BP) in a choledochal cyst. Papillomatosis arising from the intrahepatic portion of a choledochal cyst presenting with haemobilia has not been reported before. Case historyA 57-year-old woman presented initially with periumbilical pain that had lasted for a week. She also complained of tiredness but did not have loss of weight or appetite. Imaging in another hospital had revealed common bile duct thickening with intrahepatic biliary radicle dilatation and biliary sludge. She was suspected of having cholangiocarcinoma and underwent endoscopic retrograde cholangiopancreaticography (ERCP) with stent placement.General and systemic examination and blood tests were non-contributory. Magnetic resonance cholangiopancreaticography with computed tomography of the abdomen (MRCT) showed dilatation of the common bile duct and left hepatic duct (LHD). There were multiple hyperdense lesions in the LHD suggestive of intrahepatic stones (Figs 1 and 2).The patient was offered cholecystectomy, cholangioscopy, extraction of the hepatolithiasis and hepaticojejunostomy. She came back for the operation four months later. She was pale with a history of two episodes of melaena. Her haemoglobin was 5.7g% and her blood picture was consistent with iron deficiency anaemia. The coagulation profile was normal. Oesophagogastroduodenoscopy, colonoscopy and review of MRCT of the abdomen did not reveal the source of bleeding. She was diagnosed with obscure gastrointestinal bleeding and on-table enteroscopy was added to the plan of operation.

A case with massive hemobilia long-term after internal drainage surgery for congenital biliary dilation

“…The other report reviewed hemobilia from benign biliary papillomatosis in a 57-year-old woman. Division surgery was performed, and the patient was followed up uneventfully for 4 years after surgery [ 22 ]. Our case which exhibited hemobilia in the CBD patient had been performed IDS is the first report in the world literature.…”

Minor Papillary Relief in a Type IA Choledochal Cyst: A Case Report and Review of the Literature

“…Differential diagnosis includes biliary atresia that must be ruled out in neonates as a cause of obstructive jaundice, as well as lithiasis in any part of the biliary tract [7]. Biliary papillomatosis can present with a similar triad of symptoms, but is usually accompanied by gastrointestinal hemorrhage or hemobilia [17]. Pancreatic pseudocyst can be difficult to distinguish from CC.…”

Nontraumatic Hemobilia in Children

“…The most commonly reported causes of nontraumatic hemobilia in children are infection (liver abscess, 3 4 parasitic infestation [ascariasis], 5 6 anatomical abnormalities [gastric duplication 7 ], biliary pathology [papillomatosis of the gallbladder, 8 gallbladder polyps 9 ) and bleeding disorders (von Willebrand disease). 10 Other documented nontraumatic causes that have been described in adults include tumors (malignant 11 12 or benign 13 ), systemic lupus erythematosus, 14 sarcoidosis, 15 biliary pathology (cholelithiasis, cholecystitis, choledochal cyst, 16 gallbladder ulcer 17 ), pancreatitis, and warfarin therapy ( Table 1 ).…”