Tears of blood: an unusual case of haemolacria due to conjunctival vascular malformation successfully managed by embolisation.

A boy in his mid-teens presented with progressively increasing bleeding from the right eye and nostril intermittently over a period of 6 weeks. A complete ophthalmic examination revealed nothing significant. His otorhinological examination and haematological investigations were within normal limits. The patient came a month later with frank bleeding from the right eye. Ophthalmic examination revealed hyperaemia and maceration of the right lower palpebral conjunctiva. A histopathological examination of conjunctival scrapings from the site showed abnormal dilated blood vessels suggestive of a vascular malformation. Digital subtraction angiography confirmed the presence of a conjunctival micro arteriovenous malformation supplied by the external carotid and ophthalmic artery branches. He underwent successful transarterial Onyx embolisation resulting in complete resolution of the haemolacria.

Successful Endovascular Management of Pseudoaneurysm following Transarterial Chemoembolization: A Case Report.

Background and Objectives: Transarterial chemoembolization (TACE) is a widely accepted treatment for hepatocellular carcinoma (HCC). Regarding TACE, arterial injuries, such as hepatic artery spasm or dissection, can also occur, although pseudoaneurysms are rare. We report a case of pseudoaneurysm following TACE. Materials and Methods: A 78-year-old man had been undergoing TACE for HCC in segment 8 of the liver for the past 5 years, with the most recent TACE procedure performed approximately 1 month prior. He presented to the emergency department with melena that persisted for 5 days. Computed tomography revealed a pseudoaneurysm in the S8 hepatic artery with hemobilia. Results: the pseudoaneurysm was successfully treated by N-Butyl-cyanoacrylate glue embolization. Conclusions: In patients that have undergone TACE presenting with melena and hemobilia identified on CT, consideration of hepatic artery pseudoaneurysm is crucial. Such cases can be safely and effectively treated with endovascular managements.

A rare incidence of a hepatic artery pseudoaneurysm following plastic biliary stent insertion.

Hepatic artery pseudoaneurysms have been reported to occur in approximately 1% of cases after metal stenting for malignant biliary obstruction. In contrast, only a few cases have been reported as complications after plastic stenting for benign biliary disease. We report a 61-year-old man with cholangitis who presented with a rare complication of hemobilia after implantation of 7 Fr double pigtail plastic biliary stents. No bleeding was observed approximately one month after biliary stent tube removal. Contrast-enhanced CT scan revealed a circularly enhanced lesion (5 mm in diameter) in the arterial phase at the tip of the previously inserted plastic bile duct stent. Color Doppler ultrasonography enhanced the lesion and detected arterial blood flow inside. He was diagnosed with a hepatic artery pseudoaneurysm. However, he had no risk factors such as prolonged catheterization, severe cholangitis, liver abscess, or long-term steroid use. Superselective transarterial embolization using two metal microcoils was successfully completed without damage to the surrounding liver parenchyma. If hemobilia is suspected after insertion of a plastic bile duct stent, immediate monitoring using contrast-enhanced computed tomography or Doppler ultrasonography is recommended.

Multidisciplinary management of high-grade pediatric liver injuries.

OBJECTIVE: To present our experience of multidisciplinary management of high-grade pediatric liver injuries. INTRODUCTION: Pediatric high-grade liver injuries pose significant challenge to management due to associated morbidity and mortality. Emergency surgical intervention to control hemorrhage and biliary leak in these patients is usually suboptimal. Conservative management in selected high-grade liver injuries is now becoming standard of care. Management of hemobilia due to pseudoaneurysm formation and traumatic bile leaks requires multidisciplinary management. METHODS: A retrospective review was undertaken for patients presenting with blunt liver injuries at two tertiary care centers in Karachi, Pakistan, from March 2021 to December 2022. Twenty-eight patients were identified, and four patients fulfilled the criteria for grade 4 and above blunt liver injury during this period. RESULTS: One case with grade 4 liver injury developed hemobilia on 7th day of injury. He required two settings of angioembolization but had recurrent leak from pseudoaneurysm. He ultimately needed right hepatic artery ligation. Second patient presented with massive biliary peritonitis 2 days following injury. He was managed initially with tube laparostomy followed by ERCP and stent placement. The third patient developed large hemoperitoneum managed conservatively. One case with grade 5 injury expired during emergency surgery. CONCLUSION: Conservative management of advanced liver injuries can result in significant morbidity and mortality due to high risk of complications. Trauma surgeons need to have multidisciplinary team for management of these patients to gain optimal outcome.

Late-onset hemobilia due to pseudoaneurysm rupture after endoscopic ultrasound-guided hepaticogastrostomy.

We report the case of a 65-year-old man who experienced hemobilia due to rupture of a pseudoaneurysm of the left hepatic artery after endoscopic ultrasound-guided hepaticogastrostomy (EUS-HGS). The patient was diagnosed with pancreatic cancer and underwent endoscopic retrograde cholangiopancreatography for obstructive jaundice. Biliary drainage was converted to EUS-HGS due to tumor invasion in the superior duodenal angle. A partially covered metal stent was placed in the B3 intrahepatic bile duct. The procedure was completed without early complications, but 50 days later, the patient developed fever, elevated hepatobiliary enzymes, and shock. Contrast-enhanced computed tomography (CT) showed that the hepatic end of the HGS stent had moved slightly toward the stomach compared to the previous CT. A 6-mm pseudoaneurysm was also observed near the A3 and A4 branches of the left hepatic artery, coinciding with the hepatic end of the EUS-HGS stent. Hemostasis was achieved with coil embolization. Biliary hemorrhage due to rupture of a pseudoaneurysm should be considered in the differential diagnosis of biliary obstruction accompanied by bleeding after EUS-HGS.

[A Case of Hepatocellular Carcinoma with Haemobilia after Transcatheter Hepatic Arterial Embolization for Tumor Rupture].

A 79-year-old man was scheduled for surgery for hepatocellular carcinoma(HCC)after transcatheter hepatic arterial embolization for rupture. Two weeks before surgery, the patient came to our hospital with a chief complaint of back pain. First, we performed biliary drainage, under the diagnosis of HCC with obstructive jaundice due to haemobilia. Hepatectomy was performed when the patient's condition stabilized. It should be kept in mind that haemobilia may occur after TAE for HCC with bile duct tumor thrombus, and appropriate treatment should be performed when bleeding occurs.

Delayed Post-Traumatic Hemobilia in a Patient With Blunt Abdominal Trauma: A Case Report and Review of the Literature.

Hemobilia is a rare condition defined as bleeding in the biliary tract. The clinical presentation is variable. The typical manifestation consists of jaundice, upper gastrointestinal bleeding, and right upper quadrant abdominal pain. This set of symptoms is known as "Quincke's triad." It is present in only 22%-35% of cases. Post-traumatic hemobilia is an extraordinarily rare condition occurring in only 6% of the patients with hemobilia. In general, it occurs in less than 0.2% of patients with liver trauma. A delay in the development of bleeding after liver trauma is frequent. Early diagnosis is essential because massive bleeding into the biliary tract is a potentially life-threatening condition. We present a case of a patient with massive hemobilia developed 12 days after blunt abdominal trauma. Computed tomography angiography showed two pseudoaneurysms in hepatic segments V and VIII with contrast medium extravasation. We successfully performed digital subtraction angiography with selective transcatheter arterial embolization of the leaking segment VIII pseudoaneurysm. Embolization of the pseudoaneurysm in segment V was technically impracticable. Our article provides a review of the published literature focussing on the prevalence, diagnostics, and treatment of post-traumatic hemobilia.

Rapid identification of source of delayed hemobilia following endoscopic metallic stenting for malignant biliary obstruction.

Delayed hemobilia, a rare but potentially fatal complication of endoscopic metallic stenting for malignant biliary obstruction, requires prompt identification of the source of bleeding and subsequent embolization. However, hemobilia is characteristically intermittent, and computed tomography (CT) often fails to show pseudoaneurysms or extravasations. In particular, because the posterior superior pancreaticoduodenal artery (PSPDA) runs alongside the common bile duct for its whole length, it is readily obscured by metallic artifacts in that duct, such as stents, making identification of the source of bleeding by CT difficult. We have encountered three patients with delayed hemobilia from the PSPDA following endoscopic biliary stenting for malignant biliary obstruction in whom no extravasation or pseudoaneurysms were detected by contrast-enhanced CT during bleeding. However, when we identified that the PSPDA had a smaller diameter than in previous CTs in all three cases, we suspected that the PSPDA was the source of the bleeding. No extravasation or pseudoaneurysms were detected with celiac arteriography or superior mesenteric arteriography; however, extravasation and pseudoaneurysms were detected by direct PSPDA angiography. Hemostasis was achieved through embolization. Detecting a large decrease in the diameter of the PSPDA on contrast-enhanced CT during biliary bleeding may help to identify the source of that bleeding.

Two distinct episodes of life-threatening hemobilia due to a lesion of common bile duct and delayed intrapancreatic arteriobiliary fistula managed by emergency pancreatoduodenal resection.

Hemobilia is an extremely rare cause of upper gastrointestinal bleeding. It often has intermittent manifestation, which may lead to significant diagnostic delay. In 65% of the cases, the causes are iatrogenic, in 7% the cause is malignancy, in 5% - gallstones, in 8% it is inflammation (cholecystitis, parasites, reflux cholangitis), vascular abnormality is the cause in 7% (most commonly pseudoaneurysm of the hepatic artery), and pancreatic pseudocyst causes hemobilia in 1%. In almost all cases, the bleeding originates from intrahepatic or extrahepatic bile ducts, and rarely from the pancreas.

Hemobilia due to porto-biliary fistula complicating endoscopic retrograde cholangiopancreatography after a recent liver transplantation.

A 60-year-old woman with autoimmune hepatitis submitted to liver transplantation presented with a biliary anastomotic stenosis. An endoscopic retrograde cholangiopancreatography (ERCP) was complicated with a porto-biliary fistula due to the misplacement of a biliary stent. After multidisciplinary discussion, and the stent was endoscopically removed while a percutaneous transhepatic fully-covered self-expanded metal stent was placed in portal vein. Iatrogenic porto-biliary fistula following biliary stent placement is a rare and potentially life-threatening ERCP complication. In a suspected stent-related portal vein injury, this multidisciplinary strategy combining gastroenterology and radiology proved to be an effective and safe minimally invasive technique avoiding catastrophic consequences.

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.

A case of hemobilia caused by pancreatic metastasis of renal cell carcinoma treated with a covered metallic stent.

We present the case of an 86-year-old man who had undergone left nephrectomy for renal cell carcinoma (clear cell carcinoma) 22 years ago. He visited the emergency department complaining of right hypochondrial pain and fever. He was eventually diagnosed with acute cholangitis. Abdominal contrast-enhanced computed tomography showed multiple tumors in the pancreas. The tumor in the pancreatic head obstructed the distal bile duct. Endoscopic retrograde cholangiopancreatography detected bloody bile juice flowing from the papilla of Vater. Therefore, he was diagnosed with hemobilia. Cholangiography showed extrinsic compression of the distal bile duct; a 6 Fr endoscopic nasobiliary drainage tube was placed. Endoscopic ultrasound showed that the pancreas contained multiple well-defined hypoechoic masses. Endoscopic ultrasound-guided fine-needle aspiration was performed using a 22 G needle. Pathological examination revealed clear cell carcinoma, and the final diagnosis was pancreatic metastasis of renal cell carcinoma (RCC) causing hemobilia. A partially covered metallic stent was placed in the distal bile duct. Consequently, hemobilia and cholangitis were resolved.

Transcatheter arterial embolization for massive hemobilia with N-butyl cyanoacrylate (NBCA) Glubran 2.

BACKGROUND: Massive hemobilia is a life-threatening condition and therapeutic challenge. Few studies have demonstrated the use of N-butyl cyanoacrylate (NBCA) for massive hemobilia. PURPOSE: To investigate the efficacy and safety of transcatheter arterial embolization (TAE) using NBCA Glubran 2 for massive hemobilia. MATERIAL AND METHODS: Between January 2012 and December 2019, the data of 26 patients (mean age 63.4 ± 12.6 years) with massive hemobilia were retrospectively evaluated for TAE using NBCA. The patients' baseline characteristics, severities of hemobilia, and imaging findings were collected. Emergent TAE was performed using 1:2-1:4 mixtures of NBCA and ethiodized oil. Technical success, clinical success, procedure-related complications, and follow-up outcomes were assessed. RESULTS: Pre-procedure arteriography demonstrated injuries to the right hepatic artery (n = 24) and cystic artery (n = 2). Initial coil embolization distal to the lesions was required in 5 (19.2%) patients to control high blood flow and prevent end-organ damage. After a mean treatment time of 11.2 ± 5.3 min, technical success was achieved in 100% of the patients without non-target embolization and catheter adhesion. Clinical success was achieved in 25 (96.2%) patients. Major complications were noted in 1 (3.8%) patient with gallbladder necrosis. During a median follow-up time of 16.5 months (range 3-24 months), two patients died due to carcinomas, whereas none of the patients experienced recurrent hemobilia, embolic material migration, or post-embolization complications. CONCLUSION: NBCA embolization for massive hemobilia is associated with rapid and effective hemostasis, as well as few major complications. This treatment modality may be a promising alternative to coil embolization.

Challenges in Diagnosis and Management of Hemobilia.

Hemobilia, or hemorrhage within the biliary system, is an uncommon form of upper gastrointestinal (GI) bleeding that presents unique diagnostic and therapeutic challenges. Most cases are the result of iatrogenic trauma, although accidental trauma and a variety of inflammatory, infectious, and neoplastic processes have also been implicated. Timely diagnosis can often be difficult, as the classic triad of upper GI hemorrhage, biliary colic, and jaundice is present in a minority of cases, and there may be considerable delay in the onset of bleeding after the initial injury. Therefore, the radiologist must maintain a high index of suspicion for this condition and be attuned to its imaging characteristics across a variety of modalities. CT is the first-line diagnostic modality in evaluation of hemobilia, while catheter angiography and endoscopy play vital and complementary roles in both diagnosis and treatment. The authors review the clinical manifestations and multimodality imaging features of hemobilia, describe the wide variety of underlying causes, and highlight key management considerations.©RSNA, 2021.

Hemobilia due to Hepatic artery pseudoaneurysm secondary to collateral circulation formation after liver trauma: a case report.

BACKGROUND: Hemobilia due to rupture of hepatic artery pseudoaneurysm and recurrent hemorrhage caused by hepatic artery collateral circulation are both rare complications after liver trauma. There have been a number of separate reports of both complications, but no cases have been reported in which the two events occurred in the same patient. Here we report a recurrent hemorrhage in the bile duct due to hepatic artery pseudoaneurysm secondary to collateral circulation formation after hepatic artery ligation in a patient with liver trauma. CASE PRESENTATION: A 52-year-old male patient was admitted to our hospital for liver trauma (Grade IV according to the American Association for the Surgery of Trauma (AAST) grading system) with active bleeding after a traffic accident. Hepatic artery ligation was performed for hemostasis. Three months after the surgery, the patient was readmitted for melena and subsequent hematemesis. Selective angiography examination revealed the formation of collateral circulation between the superior mesenteric artery and right hepatic artery. Moreover, a ruptured hepatic artery pseudoaneurysm was observed and transcatheter arterial embolization (TAE) was performed for hemostasis at the same time. After the treatment, the patient recovered very well and had an uneventful prognosis until the last follow-up. CONCLUSION: For patients with hepatic trauma, the selection of the site of hepatic artery ligation and the diagnosis and treatment methods of postoperative biliary hemorrhage are crucial for the prognosis of the disease.

Hemobilia as a Complication of Transhepatic Percutaneous Biliary Drainage: a Rare Indication for Laparoscopic Common Bile Duct Exploration.

BACKGROUND: Hemobilia is the presence of blood in the biliary tree and is a frequent complication after percutaneous transhepatic biliary drainage (PTBD).1 Most of these episodes are self-limited; nevertheless, in less than 5% of cases, hemobilia is clinically significant, requiring an intervention (hepatic artery embolization, stenting, or percutaneous thrombin injection).2,3 Adequate treatment requires control of hemorrhage and restoration of bile flow. Surgery is the last resort and is indicated when the other modalities fail. METHODS: A 65-year-old man with multiple comorbidities was admitted with cholangitis. The patient underwent PTBD (Figure 1) but had persistent cholestasis. Thus, he underwent endoscopic cholangiopancreatography (ERCP), in which a plastic stent was misplaced within the common bile duct (CBD) and could not be removed (Figure 2). Afterwards, as the patient had persistently high bilirubin levels and the previously placed stent was malpositioned, the decision was made to proceed with laparoscopic cholecystectomy and CBD exploration. RESULTS: The operation was performed with choledocoscope guidance, and the CBD was closed over a T-tube. The operative time was 280 min. Postoperative course was uneventful; the T-tube was clamped 1 week after discharge. Four weeks postoperatively, the T-tube cholangiogram showed a patent extrahepatic biliary tree with no filling defects (Figure 3). The T-tube was then removed. CONCLUSIONS: Biliary obstruction secondary to hemobilia is a rare occurrence after PTBD. Surgical CBD exploration is required when conservative management and endoscopic treatment fail and can be done successfully through a minimally invasive approach.

Haemobilia: secondary to micro aneurysms of hepatic artery.

Hepatic artery is the fourth most common site of the intraabdominal aneurysm, after infra renal aorta, iliac artery and splenic artery aneurysms. Rupture of the aneurysm may lead to the upper gastrointestinal haemorrhage. Here we report a 5 years old boy, who presented with fever, abdominal distension and unexplained upper GI bleed. Upper GI endoscopy revealed a normal esophagus and stomach with clear evidence of haemobilia with blood oozing from the ampulla. Fluoro- guided angiography followed by embolization of hepatic artery branches with 5 metallic coils was performed in this case by an interventional radiologist.