Massive hemobilia and papillomatosis of the gallbladder in metachromatic leukodystrophy: a life-threatening condition.

Polyposis of the gallbladder is rare during childhood. This condition can be associated with three other conditions: metachromatic leukodystrophy, Peutz-Jeghers' syndrome, and pancreaticobiliary maljunction. We report the case of a child with hemobilia in metachromatic leukodystrophy, which rendered cholecystectomy necessary. Macroscopically, the gallbladder measured 4.6 cm in length and showed an opaque serous surface and focal brown petechiae. Moreover, a yellow polypoid lesion of 2 cm in diameter and a diffuse thickening of the fundus wall were observed. Many reports describe the importance of the association of gallbladder papillomatosis with metachromatic leukodystrophy, but only three cases presented with massive intestinal bleeding, such as our young patient had. It is thus imperative that this life-threatening condition should be well known.

[Hemobilia from pancreatic arteriovenous malformation: successful treatment with transportal coil embolization of draining veins].

Pancreatic arteriovenous malformations (AVM) are extremely rare diseases frequently complicated by gastrointestinal hemorrhage. While surgical resection of affected lesion is preferred for the treatment of pancreatic AVM, angiographic intervention can be used as an alternative treatment, especially in surgically high-risk patients. We experienced a patient with pancreatic AVM manifested by hemobilia and biliary sepsis. Superior mesenteric and common hepatic arteriography showed pancreaticoduodenal AVM composed of nidus supplied by numerous fine feeding arteries and of draining veins encircling the common bile duct (CBD). Hemobilia was controlled by transportal coil embolization of draining veins of AVM around the CBD. Herein, we report this case with the review of literatures.

Percutaneous liver biopsy complicated by hemobilia-associated acute cholecystitis.

Liver biopsy is generally considered a safe and highly useful procedure. It is frequently performed in an outpatient setting for diagnosis and follow-up in numerous liver disorders. Since its introduction at the end of the 19th century, broad experience, new imaging techniques and special needles have significantly reduced the rate of complications associated with liver biopsy. Known complications of percutaneous biopsy of the liver include hemoperitoneum, subcapsular hematoma, hypotension, pneumothorax and sepsis. Other intra-abdominal complications are less common. Hemobilia due to arterio-biliary duct fistula has been described, which has only rarely been clinically expressed as cholecystitis or pancreatitis. We report a case of a fifteen year-old boy who developed severe acute cholecystitis twelve days after a percutaneous liver biopsy performed in an outpatient setting. The etiology was clearly demonstrated to be hemobilia-associated, and the clinical course required the performance of a laparoscopic cholecystectomy. The post operative course was uneventful and the patient was discharged home. Percutaneous liver biopsy is a safe and commonly performed procedure. However, severe complications can occasionally occur. Both medical and surgical options should be evaluated while dealing with these rare incidents.

Haematochezia in Crohn's disease caused by late-onset haemobilia following percutaneous liver biopsy.

The case of a patient with Crohn's disease and who presented 4 days after a percutaneous liver biopsy with haematochezia suggestive of an exacerbation of his inflammatory bowel disease is described. Subsequent abdominal ultrasonography revealed free fluid in the peritoneal cavity and around the gallbladder. Haemobilia was suspected. Selective hepatic arteriography demonstrated a fistula between the hepatic artery and the portal venous system, with the presence of a hepatic artery pseudoaneurysm. Bleeding was stopped by arterial embolisation using titanium microcoils. Haemobilia is a very rare complication of percutaneous liver biopsy, but it should not be overlooked as a potential late-onset cause of rectal bleeding following this procedure. Selective hepatic arterial embolisation is an effective and safe treatment for this serious complication.

Heterotopic gastric mucosa in intrahepatic bile duct, presenting with hemobilia: a case report.

We present a 66-year-old man with unique heterotopic gastric mucosa in the intrahepatic bile duct causing hemobilia. Endoscopic retrograde cholangiography showed irregular stenosis of the left intrahepatic bile duct, and a provisional diagnosis of cholangiocarcinoma was made. Therefore, partial hepatic lobectomy and cholecystectomy were performed. Histological examination of the liver showed the presence of ectopic gastric mucosa in the intrahepatic bile duct containing mucous glands with parietal and chief cells and bile. Heterotopic gastric mucosa in the intrahepatic bile duct is a rare cause of hemobilia.

Pancreaticobiliary arteriovenous malformation with common bile duct dilation in a patient with hemobilia.

Arteriovenous malformation of the pancreas is a very rare entity. We describe a case involving the head of the pancreas associated with progressive hemobilia bleeding from the lower part of the common bile duct. The patient was a 50-year-old man with acute epigastralgia. Endoscopic retrograde cholangiopancreatography revealed hemobilia and cystic dilation of the common bile duct. Angiography demonstrated increased blood volume in the head of the pancreas and early filling of the superior mesenteric and portal veins. Abdominal pain and progressive anemia caused by hemobilia required surgical treatment. Histologic examination of the resected specimen revealed marked proliferation of the blood vessels in the pericholedochal area and the exact point of bleeding from the pancreaticobiliary arteriovenous malformation.

Cholecystitis caused by hemocholecyst from underlying malignancy.

Massive hemobilia is a well recognized clinical entity, particularly when it presents with jaundice, GI bleeding, and biliary pain. However, occult hemobilia is more difficult to diagnose and has seldom been reported because of its clinically silent nature. In fact, this is usually overlooked until complications arise. Hemocholecyst or clot within the gallbladder may rarely occur in this setting, leading to cystic duct obstruction and cholecystitis. Most previous reports describe cholecystitis resulting from hemocholecyst after iatrogenic trauma. We describe two cases in which hemocholecyst occurred from underlying malignancies, both resulting in cholecystitis (acute or chronic).

Hemobilia caused by a giant benign hemangioma of the liver: report of a case.

We report herein the extremely unusual case of a 39-year-old woman in whom a giant cavernous hemangioma caused hemobilia. Cavernous hemangioma is the most common benign neoplasm of the liver and rarely causes any clinical symptoms or signs, while hemobilia usually occurs secondary to accidental operative or iatrogenic trauma, vascular disease, inflammatory disorders, gallstones, or tumors of the liver. Although invasive or malignant hepatic tumors often result in a communication between the biliary tract and the blood vessels, only one case of hemobilia caused by a benign cavernous hemangioma has ever been reported, but with no details about the patient. Our patient presented to a local hospital with severe melena as the initial main symptom, where ligation of the right hepatic artery was performed. This failed to relieve her symptoms, and she was subsequently referred to our department where a right hepatectomy was performed. Histopathological examination revealed no malignancy combined with the tumor; however, the hemangioma was exposed to the bile duct in segment VIII, which was presumably the cause of the hemobilia. This patient remains in good health almost 6 years after her operation. To the best of our knowledge this is the first case report of hemobilia caused by a cavernous hemangioma, and is accompanied by a detailed analysis.

[Aneurysm of the cystic artery as a rare cause of hemobilia]. / Aneurysma der Arteria cystica als seltene Ursache einer Hämobilie.

A 52-year old woman was admitted to the hospital because of upper abdominal pain and hematemesis. Laboratory parameters showed marked cholestasis. Endoscopic retrograde cholangiopancreatography (ERCP) lead to the diagnosis of hemobilia. CT-scan and angiography revealed an aneurysm of the cystic artery as the cause of hemobilia. Cholecystectomy was performed because of concomitant cholecystitis. Anatomical examination confirmed clinical diagnosis.

Poly (2-hydroxyethyl methacrylate) particles for management of hemorrhage of complicated origin: treatment of hemobilia.

Poly (2-hydroxyethyl methacrylate) (PHEMA) particles of cylindrical and spherical shape were developed as a preparation for tumor treatment or control of hemorrhage by blocking their blood supply. In this report, PHEMA particles were used for the management of hemobilia, that is, bleeding into biliary passages. The origin of hemobilia in 31 patients was localized by selective angiography. With the objective of prophylaxy of hemorrhage, selective embolization with PHEMA particles of the branches of the hepatic artery responsible for the supply of blood to the focus of damage was used in 18 patients. This low-trauma method allowed either a complete control of bleeding or, at least, intraoperative blood loss was reduced more than twice. Histological investigation of the occluded blood vessels showed that the thrombus was attached to the particles and was reinforced by the porous structure of the polymer. A hypercoagulation reaction was observed in the postembolization period. This allowed correction of the hypocoagulation in the hemostasis system.

Massive hemobilia and acalculous cholecystitis due to benign gallbladder polyp.

A 37-year-old male with metachromatic leukodystrophy, a congenital cerebroside storage disease strongly associated with benign gallbladder polyps, presented with hemobilia and acalculous cholecystitis due to a long, slender, benign gallbladder polyp. This case report extends the clinical spectrum of gallbladder polyps and demonstrates a novel cause of hemobilia. The unusually long, slender polyp shape may have promoted hemorrhage due to the potential for torsion around a slender stalk. The mechanism of cholecystitis may have been cystic duct obstruction from blood clots or possibly from prolapse by the long, slender polyp into the cystic duct.

Hemobilia: presentación clínica y diagnóstico / Hemobilia: clinical presentation and diagnosis

La hemobilia es una rara causa de hemorragia digestiva superior, que requiere de su sospecha para hacer un diagnóstico rápiso y preciso, que permita reducir su alto índice de mortalidad. Presentamos nuestra experiencia en tres casos, analizando las manifestaciones clínicas, los métodos diagnósticos y evolución de los mismos

Hemobilia from ruptured hepatic artery aneurysm: angiographic demonstration of arteriobiliary fistula in a successfully treated case.

Rupture of a hepatic artery aneurysm into the biliary tree is a rare cause of hemobilia. The dramatic nature of the disease and the difficulties in obtaining a preoperative diagnosis are the main causes of its high mortality rate. Progress in vascular radiology and surgery seems to be an important factor in improving the survival rates in patients with this type of hemobilia. A case of a patient with an aneurysm of the common hepatic artery ruptured into the common bile duct is reported. Preoperative arteriography revealed the aneurysm and the arteriobiliary fistula causing massive hemobilia. The patient was successfully treated by arterial ligation and is symptom free after 2 years.