Peroral cholangioscopic diagnosis of refractory hemobilia caused by pancreatic arteriovenous malformation: a case report
Min Xu, Yingying Li, Yanru Li, Yuwei Wang, Shuyi Zhang, Wen Li, Hao Zhang
Abstract
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TopicsAbdominal vascular conditions and treatments · Pancreatitis Pathology and Treatment · Gallbladder and Bile Duct Disorders
Pancreatic arteriovenous malformation is a rare vascular anomaly. The first reported case was described in 1968 by Halpern et al. 1 . Yang Zhuo et al. reported a case of failed treatment of biliary hemorrhage caused by a gastroduodenal artery malformation with fully covered biliary metal stents 2 . In this case, the patient was admitted due to “common bile duct stones” and experienced hemobilia after endoscopic retrograde cholangiopancreatography (ERCP). Peroral cholangioscopy revealed bleeding from a cystic lesion in the pancreatic portion of the common bile duct.
A 67-year-old man was admitted to our hospital with upper abdominal pain. Magnetic resonance cholangiopancreatography showed an abnormal signal intensity in the pancreatic head, common bile duct stones and pancreatitis ( Fig. 1 ). The patient underwent ERCP to remove the stone ( Fig. 2 a–d ). The patient developed abdominal pain, blood in the nasobiliary drainage, and melena after ERCP. No active bleeding was observed at the endoscopic sphincterotomy incision margin ( Fig. 2 e–f ). Peroral cholangioscopy revealed a cystic dilation of the distal common bile duct. Within the cyst, the normal bile duct wall architecture was absent, and tortuous, dilated vessels along with a hematoma were observed ( Fig. 2 g–h ). The cholangioscopic findings suggested bleeding from a vascular malformation ( Video 1 ).
a, b MRCP revealing cystic dilation of the pancreatic segment of the common bile duct. c MRCP reveals a hypointense focus within the common bile duct. d Abdominal CT revealing dilation of the pancreatic segment containing hyperdensity. CT, computed tomography; MRCP, magnetic resonance cholangiopancreatography
The first ERCP: a hemobilia from the orifice of Vater’s ampulla before cannulation. b ERCP reveals dilation of the extrahepatic bile duct within stones and cystic dilation of the pancreatic segment of the common bile duct. c Bloody bile drainage is observed after stone removal. d Fluoroscopy reveals a blood clot within the cystic dilation. e No active bleeding was observed at the EST incision margin. The second ERCP: f fluoroscopy reveals a blood clot within the cystic dilation. g, h Peroral cholangioscopy revealed a cystic dilation of the distal common bile duct. Within the cyst, the normal bile duct wall architecture was absent, and tortuous, dilated vessels along with a hematoma were observed. ERCP, endoscopic retrograde cholangiopancreatography; EST, endoscopic sphincterotomy.
Peroral cholangioscopy shows a cystic dilation in the distal common bile duct with a hematoma. During DSA, the pancreatic head region exhibits dense vascularity, accompanied by rapid visualization of the portal vein. DSA, digital subtraction angiography.Video 1
During digital subtraction angiography (DSA), angiographic findings were considered typical of an arteriovenous malformation in the pancreaticobiliary region ( Fig. 3 a ). After the superior pancreatoduodenal artery was embolized, post-embolization angiography confirmed the disappearance of the vascular cluster and the fistula ( Fig. 3 b , Video 1 ). Subsequently, the hemobilia occurred again. An abdominal contrast-enhanced computed tomographic scan showed that the surgical intervention was not feasible ( Fig. 4 a, b ). A repeat DSA was performed, and the superior pancreaticoduodenal artery was embolized using coils ( Fig. 4 c, d ). No further bleeding occurred following this procedure. The nasobiliary tube was removed 7 days later. Unfortunately, the patient died due to a cardiocerebrovascular accident.
Enhanced computed tomography reveals multiple hypervascular spots in the pancreatic head and early visualization of the portal vein and superior mesenteric vein.
The first DSA: a GDA angiography: the pancreatic head region exhibits dense vascularity, accompanied by rapid visualization of the portal vein. b The arterioportal fistula vanished subsequent to the gelatin sponge embolization of the superior pancreaticoduodenal artery. The second DSA: c Angiography of the common hepatic artery still demonstrates blood supply to the pancreatic head region via the superior pancreaticoduodenal artery and d followed by coil embolization of the superior pancreaticoduodenal artery. DSA, digital subtraction angiography; GDA, gastroduodenal artery.
Endoscopy_UCTN_Code_CCL_1AZ_2AN
The reference list from the paper itself. Each links out to its DOI / PubMed record.
- 1Halpern M Turner AF Citron BP Hereditary hemorrhagic telangiectasia. An angiographic study of abdominal visceral angiodysplasias associated with gastrointestinal hemorrhage Radiology 1968901143114910.1148/90.6.11435656734 · doi ↗ · pubmed ↗
- 2Han K Zhao Q Sun Y Direct peroral cholangioscopic diagnosis and metal stent failure in a case of refractory arterial hemobilia Endoscopy 20255701 E 1096 E 109710.1055/a-2686-341440983102 PMC 12453878 · doi ↗ · pubmed ↗