Rare cause of obstructive haemobilia with recurrent biliopancreatic complications: a paradigmatic case.

Haemobilia is an unusual but significant cause of upper gastrointestinal bleeding. Two-thirds of haemobilia cases are secondary to invasive hepato-biliopancreatic procedures. Biliary angiodysplasia is exceptionally unusual, with only three cases reported. Herein, we report the case of an autonomous 80-year-old woman with a history of cholecystectomy 5 years ago and cardiovascular disease-hypertension, heart failure, acute myocardial infarction, stroke and non-valvular atrial fibrillation, anticoagulated with apixaban 2.5 mg two times per day. Since July 2019, she had four episodes of acute cholangitis of mild-to-moderate severity, having undergone broad spectrum antibiotics treatment and endoscopic retrograde cholangiopancreatography (ERCP), with sphincterotomy and bile sludge extraction. After 3 months, the patient presented with a new episode of acute cholangitis, this time with haemobilia (Quincke's triad). An abdominal CT angiography showed no evidence of active bleeding, with plastic biliary prosthesis left by ERCP. The patient continued presenting new episodes of acute cholangitis with haemobilia, some of them with associated pancreatitis. A cholangioscopy with Spyglass DS II was performed, showing an angiodysplasia occupying half of the luminal circumference of the middle choledoccus, without active haemorrhage. After a multidisciplinary meeting and given the high haemorrhagic/thrombotic risk (CHA2DS2-VASc 8), closure of the left atrial appendage was considered. However, relapse of the condition after beginning the antiaggregation protocol for cardiovascular intervention made it unfeasible. Another cholangioscopy with an ultra-thin endoscope for argon-plasma coagulation was attempted, without success. The abdominal CT angiography was repeated, this time with identification of dilated ramifications of the gastroduodenal and inferior pancreatic arteries. After embolisation of these aberrant vessels with microcoils, the patient went well, with no recurrence of bleeding or biliopancreatic complications. We present a case of obstructive haemobilia with multiple biliopancreatic complications, secondary to an extremely rare cause-choledochal angiodysplasia. Cholangioscopy had a decisive role in the diagnosis and therapeutic guidance. The diagnostic/therapeutic challenge associated with haemobilia stands out, with the need for a personalised and multidisciplinary approach.

Hemobilia and acute hemocolecyst as an unusual presentation of gallbladder cancer.

Gallbladder cancer that presents as acute cholecystitis associated with hemocholecyst and hemobilia is a rare entity. There are few cases reported in the literature. We present a case of gallbladder carcinoma diagnosed after emergency cholecystectomy, performed due to hemobilia and acute cholecystitis due to hemocholecyst.

El cáncer de vesícula biliar que se presenta como una colecistitis aguda asociada a hemocolecisto y hemobilia es muy infrecuente. Hasta la fecha hay pocos casos informados en la literatura. Presentamos un caso de carcinoma de vesícula biliar diagnosticado tras colecistectomía de urgencia, realizada por hemobilia y colecistitis aguda por hemocolecisto.

Haemobilia secondary to an arterio-biliary fistula: A rare complication of intra-operative microwave ablation of hepatocellular carcinoma.

A rare case of arterio-biliary fistula and haemobilia complicating intra-operative microwave ablation of hepatocellular carcinoma in a 58-year-old woman with cirrhosis.

Emergency surgery for hemobilia due to hepatic artery pseudoaneurysm rupture complicated by Mirizzi syndrome type II: a case report.

BACKGROUND: Hemobilia refers to bleeding into the biliary tract. Hepatic artery pseudoaneurysm (HAP) rupture is an uncommon cause of hemobilia, and cases of HAP associated with Mirizzi syndrome are extremely rare. Although transarterial embolization is recommended as the first-line treatment for hemobilia, surgery is sometimes required. CASE PRESENTATION: A 76-year-old woman was referred to our hospital with epigastric pain. She was febrile and had conjunctival icterus and epigastric tenderness. Laboratory tests revealed abnormal white blood cell count and liver function. An abdominal computed tomography (CT) revealed multiple calculi in the gallbladder, an incarcerated calculus in the cystic duct, and a slightly dilated common hepatic duct. Based on examination findings, she was diagnosed with Mirizzi syndrome type I, complicated by cholangitis. Intravenous antibiotics were administered, and we performed endoscopic retrograde cholangiopancreatography (ERCP) to place a drainage tube. The fever persisted; therefore, contrast-enhanced CT (CECT) was performed. This revealed portal vein thrombosis and hepatic abscesses; therefore, heparin infusion was administered. The following day, she complained of melena, and laboratory tests showed that she was anemic. ERCP was performed to change the drainage tube in the bile duct; however, bleeding from the papilla of Vater was observed. CECT demonstrated a right HAP with high-density fluid in the gallbladder and gallbladder perforation. Finally, she was diagnosed with hemobilia caused by HAP rupture, and emergency surgery was performed to secure hemostasis and control the infection. During laparotomy, we found that a right HAP had ruptured into the gallbladder. The gallbladder made a cholecystobiliary fistula, which indicated Mirizzi syndrome type II. Although we tried to repair the right hepatic artery, we later ligated it due to arterial wall vulnerability. Then, we performed subtotal cholecystectomy and inserted a T-tube into the common bile duct. There were no postoperative complications except for minor leakage from the T-tube insertion site. The patient was discharged after a total hospital stay of 7 weeks. CONCLUSIONS: We experienced an extremely rare case of emergency definitive surgery for hemobilia due to HAP rupture complicated by Mirizzi syndrome type II. Surgery might be indicated when controlling underlying infections was required.

Haemobilia secondary to a cystic artery pseudoaneurysm as complication of VLC.

INTRODUCTION: The laparoscopic approach has become the gold standard for cholecystectomy. However, it could have some major complications. Among them, it can be considered postoperative pseudoaneurysms of the cystic or hepatic arteries. Haemobilia secondary to a cystic artery pseudoaneurysm is extremely rare. CASE REPORT: Here we present a case from our centre of haemobilia in association with a cystic artery pseudoaneurysm, as a late complication of VLC. An 18-year-old girl underwent laparoscopic cholecystectomy; during surgery, due to viscero-perietal tight adhesions and due to the close proximity of the cystic duct to the biliary ducts, we suspected a bile duct injury. So, decision was taken to convert to open surgery: a suture was performed to repair the coledocic duct injury and an endoscopic papillotomy was performed with subsequent positioning of an endoscopic plastic biliary endoprothesis at the hepatocholedochus. One month after surgery, the patient showed clinical signs of hypovolemic shock. She underwent Computed Tomography Angiography, showing a possible arterial lesion, just adjacent to surgical clip. Therefore, patient underwent angiographic examination, which confirmed an 8 mm pseudoaneurysm arising from cystic artery, just adjacent to surgical clips. Superselective catheterization of vessel was performed, and two coils were released, until obtaining complete exclusion of the vascular lesion. The patient was discharged five days after procedure, with good general condition. CONCLUSION: Pseudoaneurysms of the cystic artery are uncommon entities, rarely reported in the literature, and often caused by cholecystitis or iatrogenic biliary injury. All conditions that are responsible for vessels' injuries could also cause haemobilia. Even if pseudoaneurysm of cystic artery with haemobilia is a rare event, it has to be considered as a complication of VLC. Angiographic approach should be the treatment of choice.

Revisiting Quincke's Triad: A Case of Idiopathic Hepatic Artery Aneurysm Presenting with Obstructive Jaundice.

INTRODUCTION: Hepatic artery aneurysm (HAA) is a rare occurrence. Quincke's triad of hemobilia; abdominal pain, obstructive jaundice, and upper gastrointestinal (GI) bleeding could be detected in one-third of HAA patients. CASE PRESENTATION: We present a case of HAA with all signs of Quincke's triad and shock. The diagnosis of HAA was enforced by CT angiography. An urgent open surgical approach was elected by the surgical team. The patient underwent an uneventful resection of the HAA, and primary repair of the CHA followed with bilioenteric reconstruction. CONCLUSIONS: Recognizing the signs of Quincke's triad aids in prompt diagnosis of hemobilia in HAA, which suggests a rupture of the aneurysm or fistula formation into the biliary tree that would need urgent management by both vascular and HBP surgeons.

Hemobilia after bile duct resection: perforation of pseudoaneurysm into intra-pancreatic remnant bile duct: a case report.

BACKGROUND: Hemobilia occurs mainly due to iatrogenic factors such as impairment of the right hepatic or cystic artery, and/or common bile duct in hepatobiliary-pancreatic surgery. However, little or no cases with hemobilia from the intra-pancreatic remnant bile duct after bile duct resection (BDR) has been reported. Here, we report a case of massive hemobilia due to the perforation of psuedoaneurysm of the gastroduodenal artery (GDA) to the intra-pancreatic remnant bile duct after hepatectomy with BDR. CASE PRESENTATION: A 68-year-old male underwent extended right hepatectomy with BDR for gallbladder carcinoma. He presented with upper gastrointestinal bleeding 2 months after the initial surgery. Upper endoscopy identified a blood clot from the ampulla of Vater and simultaneous endoscopic balloon tamponade contributed to temporary hemostasis. Abdominal CT and angiography revealed a perforation of the psuedoaneurysm of the GDA to the intra-pancreatic remnant bile duct resulting in massive hemobilia. Subsequent selective embolization of the pseudoaneurysm with micro-coils could achieve complete hemostasis. He survived without any recurrence of cancer and bleeding. CONCLUSION: Hemobilia could occur in a patient with BDR due to perforation of the pseudoaneurysm derived from the GDA to the intra-pancreatic remnant bile duct. Endoscopic balloon tamponade was useful for a temporal hemostasis and a subsequent radiologic interventional approach.

Triple Covered Metal Stent Deployment Using Side-by-Side Technique for Hemobilia due to Hepatocellular Carcinoma (with Video).

A hepatocellular carcinoma (HCC) rarely expands into the biliary tract. In this situation, because of its hypervascular nature, cholangitis or hemobilia may sometimes occur. Surgery is one of the options in this situation. However, patients with HCC and bile duct invasion are sometimes in a poor general condition, as in the case presented in this report. For such patients, surgical treatment may need to be invasive. Thus, here we report technical tips for triple covered metal stent deployment using side-by-side technique for hemobilia due to HCC. After guidewire deployments at the left, anterior, and posterior bile ducts, 6-mm covered self-expandable metal stents were placed at each bile duct. This may be useful for high-grade hepatic hilar obstruction due to HCC because drainage and hemostasis effects are obtained.

Spontaneous right hepatic artery branch gallbladder fistula revealed by haemobilia and upper cataclysmic gastrointestinal bleeding.

Spontaneous right hepatic artery branch gallbladder fistula is a rare condition. Our case reported a spontaneous fistula between the right branch of the hepatic artery and the gall bladder. It constitutes a rare cause of haemobilia. In fact, the most common aetiology of haemobilia is traumatic or iatrogenic secondary to hepatobiliary surgery or interventions. Diagnosis of vascular-biliary fistula is not easy. The gallbladder endoluminal clot can mimic a mass, as in our patient. Selective arterial angiography is helpful in identifying the source of gastrointestinal haemorrhage. It can demonstrate the presence of arteriobiliary fistula. The differential diagnosis is arterial pseudoaneurysm in the vicinity of the vessel. Mini-invasive treatment of this fistula constitutes the best treatment. We here report a case of haemobilia with upper cataclysmic gastrointestinal bleeding revealing a spontaneous fistula between the right branch of the hepatic artery and the gall bladder.

Recurrent Bleeding from a Hepatic Artery Pseudoaneurysm after Biliary Stent Placement.

A 78-year-old woman was admitted with benign biliary stenosis. A plastic stent was placed at the left branch to prevent obstructive cholangitis. Two weeks after the procedure, the patient was readmitted with cholangitis caused by hemobilia. However, computed tomography (CT), endoscopic retrograde cholangiopancreatography (ERCP), peroral cholangioscopy, and abdominal angiography failed to establish the bleeding source. At the seventh bleeding, CT revealed a hepatic artery pseudoaneurysm for which coil embolization was successfully performed. Hemobilia after plastic stent placement is extremely rare. We urge clinicians to consider the possibility of a pseudoaneurysm near the stent when trying to identify the bleeding source.

Ascending Cholangitis secondary to migrated embolization coil of gastroduodenal artery pseudo-aneurysm a case report.

BACKGROUND: Gastroduodenalartery (GDA) pseudo-aneurysms are very rare. Their clinical importance lies in the eventuality of rupture, causing bleeding and ultimately exsanguination. CASE PRESENTATION: We report the case of a man, with prior history of biliary surgery, presenting with haemobilia secondary to a rupture of GDA pseudo-aneurysm eroding the main bile duct. The patient was treated with coil embolization. This technique is considered to be safe. However, on the long term, some complications may occur. In our case, the patient presented with cholangitis subsequent to coil migration in the lower bile duct. This situation was managed using endoscopic retrograde cholangiopancreatography (ERCP) allowing coil extraction with favorable evolution. CONCLUSIONS: GDA pseudo-aneurysms are very rare. Bleeding, secondary to the rupture of these lesions, is a serious complication that could lead to death. Diagnosis and treatment of ruptured GDA pseudo-aneurysms rely on angiography. This method is considered to be safe. Cholangitis secondary to coil migration in the main bile duct is exceedingly rare,but remains an eventuality that physicians should be cognizant of.

Spontaneous hemobilia after liver transplantation: Frequency, risk factors, and outcome of endoscopic management.

BACKGROUND AND AIM: Spontaneous hemobilia is an uncommon liver transplantation (LT)-related biliary complication. The frequency, etiology, and mechanism of spontaneous hemobilia after LT are not known. This study aimed to assess the outcome of endoscopic management for spontaneous hemobilia after LT, and to investigate its frequency and risk factors. METHODS: The records of patients who underwent endoscopic retrograde cholangiopancreatography to manage hemobilia after LT at the Asan Medical Center, Korea, between January 2006 and April 2014 were retrospectively reviewed. RESULTS: A total 2701 cases of LT was performed in the study period, and 33 LT patients with spontaneous hemobilia were included in the study group. Endoscopic nasobiliary drainage was achieved in 33 cases (100%). In 29 of 33 patients (87.9%), hemobilia was improved. The frequency of spontaneous hemobilia was 1.22% (33/2701). On multivariate analysis, United Network for Organ Sharing status I or IIa (odds ratio [OR] 3.095, 95% CI 1.097-8.732, P = 0.033), alcoholic liver cirrhosis (OR 3.942, 95% CI 1.261-12.324, P = 0.018), and body mass index < 24.5 kg/m2 (OR 2.329, 95% CI 1.005-5.397, P = 0.049) were significant risk factors for spontaneous hemobilia after LT. CONCLUSIONS: Endoscopic retrograde cholangiopancreatography and endoscopic nasobiliary drainage are feasible methods for the management of spontaneous hemobilia after LT. In patients with United Network for Organ Sharing status I and IIa, alcoholic liver cirrhosis, or body mass index < 24.5 kg/m2 , special attention should be paid to the occurrence of spontaneous hemobilia after LT.

Angiosarcoma of the gallbladder with hemobilia: a case report.

A 61-year-old woman presented to our hospital with epigastric pain. She underwent abdominal contrast-enhanced computed tomography, which showed signal enhancement in the gallbladder fundus. As biliary obstruction was suspected, endoscopic nasobiliary drainage was performed, which revealed hemobilia. Based on this finding, gallbladder tumor was suspected, and open cholecystectomy was performed. Immunohistological staining of the resected tissue was positive for factor VIII that led to the diagnosis of gallbladder angiosarcoma. Hepatectomy and biliary reconstruction were performed for disease control; however, the patient died due to multiple liver metastases 4 months after the surgery.

Multidetector CT diagnosis of massive hemobilia due to gallbladder polyposis in a child with metachromatic leukodystrophy.

Hemobilia secondary to gallbladder polyposis is rare in children but has been reported in a few children with metachromatic leukodystrophy. We present a case with preoperative multidetector computed tomography (MDCT) diagnosis of massive hemobilia caused by gallbladder polyposis in a patient with metachromatic leukodystrophy. Our report highlights the importance of both awareness of the association of gallbladder polyposis with other syndromes such as metachromatic leukodystrophy as well as the possibility of this entity presenting with life-threatening bleeding.

Transarterial catheter embolisation for an unusual cause of upper gastrointestinal haemorrhage.

A 72-year-old man with a history of gallstones, and complex cardiac and endocrinological comorbidities, presented with severe abdominal pain and melaena. CT mesenteric angiogram showed a cystic artery pseudoaneurysm and gallbladder distended by haematoma. Subsequent mesenteric angiography confirmed a cystic artery pseudoaneurysm, which was successfully embolised with microcoils. The patient made a rapid recovery and was discharged after 3 days.

Biliary cystadenocarcinoma: an unusual cause for recurrent hemobilia.

Hemobilia is an uncommon presentation of biliary tract or pancreatic disease. The investigation and management of this clinical problem is challenging. We report on a case of biliary tract hemorrhage from an otherwise asymptomatic right lobe biliary cystadenocarcinoma and review the literature on this unusual presentation. Hemobilia from primary or secondary liver tumors is not frequently reported in the literature. Hemobilia in this setting is usually observed in association with an obvious liver mass or abnormal liver function tests. This is a report of a case of hemobilia as the primary presentation of a small right lobe cystadenocarcinoma. Literature on the incidence and treatment of hemobilia associated with liver tumors has been reviewed. Hemobilia is investigated and definitively treated with angiography. In our case, initial imaging was equivocal and the lesion was only demonstrated after rebleeding, requiring a second angiogram. Surgical resection of the mass was required for definitive control of bleeding. This case illustrates the difficulties of investigating and treating hemobilia caused by primary or secondary liver tumors. Cystadenocarcinoma of the liver is not a common tumor, and biliary tract hemorrhage as the primary presentation of this tumor in the absence of a significant mass or abnormal liver function tests has not been previously described.