biliary cast syndrome ( bcs ) is characterized by the presence of casts within the intra- or extrahepatic biliary system .
retained lithogenic material in the bile duct may lead to obstruction of the biliary tract and also to cholangitis [ 1 , 2 ] .
bcs is an uncommon complication which is mostly described in orthotopic liver transplantation [ 1 , 2 , 3 , 4 ] .
bcs is characterized by the presence of casts causing obstruction in the bile duct tree .
formation in this syndrome included exudative inflammation of the bile duct epithelium [ 6 , 7 ] , ischemic injury to the biliary endothelium , use of postoperative biliary drainage tubes and biliary infection .
bcs has also rarely been reported in non - liver transplant patients [ 9 , 10 , 11 , 12 , 13 , 14 ] .
there is no recommended standard management for patients who develop cholangiopathy secondary to bcs , and each case should be managed individually .
we describe a male long - term opium inhaler with bcs who underwent successful endoscopic removal of the cast by balloon enteroscopy - guided endoscopic retrograde cholangiopancreatography .
a 52-year - old man , who was a known case of opium addiction , presented with the chief complaint of epigastric pain for 1 week prior to admission .
routine laboratory evaluation revealed cholestatic liver enzyme elevation with alkaline phosphatase 1,810 iu / l ( reference range 80306
iu / l ) , total bilirubin 0.5 mg / dl ( reference range 0.11.2 mg / dl ) with normal aminotransferases , high erythrocyte sedimentation rate ( 90 mm / s ) and normal prothrombin time .
serum levels of fasting blood sugar , blood nitrogen urea , creatinine , triglyceride , total and low - density lipoprotein cholesterol and uric acid as well as thyroid function tests were in the normal range .
an abdominal ct scan demonstrated dilated intrahepatic and extrahepatic bile ducts ( the common bile duct measured 11 mm ) .
magnetic resonance cholangiopancreatography showed a distended gallbladder and a dilated bile duct with the conclusion of ampullary and periampullary lesion ( fig .
endoscopic retrograde cholangiopancreatography showed a linear filling defect in the intra- and extrahepatic duct ( fig .
after removal of the biliary cast he is receiving ursodeoxycholic acid ( udca ) 750 mg per day and does not report any problem 4 months after treatment .
his last laboratory evaluation revealed a significant decrease in erythrocyte sedimentation rate ( 30 mm / s ) and a mild decrease in alkaline phosphatase ( 1,423 iu / l ) .
bcs is an obstructive cholangiopathy , defined by the presence of casts within the intra- or extrahepatic biliary tract due to several mechanisms [ 1 , 2 ] .
most of the previously reported cases of bcs were described in orthotopic liver transplantation [ 1 , 2 , 3 , 4 ] . based on our knowledge and the literature
, there are only five published report of bcs in a non - transplant situation [ 9 , 10 , 11 , 12 , 13 , 14 ] .
described bcs in a patient with traumatic head injury and prolonged intensive care unit stay .
gleeson et al .   described bcs in two patients , one with hepatic infarction due to antiphospholipid syndrome and the other with fasting , parenteral nutrition and infection .
koo et al .   described a case of bcs in an elderly man after head trauma and long - term bed - ridden state .
those studies considered increased bile pigment load due to absence of gallbladder [ 9 , 12 ] , ischemic events [ 10 , 11 , 12 , 13 ] and gallbladder hypocontractility [ 11 , 14 ] as the leading causes for biliary cast formation .
structurally , in the liver transplant patients the biliary cast is composed of high bilirubin content ( 1050% ) , bile acids ( 1015% ) , few cholesterol and protein ( 510% ) or collagen from necrotic biliary epithelial cells .
however , the chemical composition of biliary casts in the setting of non - liver transplant patients is unclear .
there is no standard management for bcs patients , and a variety of therapeutic and surgical methods are used to treat bcs .
as far as we know , this is the first reported case of bcs in an opium inhaler . in our case none of the previously reported risk factors , including liver transplantation , fasting , total parenteral nutrition , hemolysis , abdominal surgery , trauma or biliary infection , was detected . in long - term opium users , bile duct size and biliary pressure
thus , we think that biliary dyskinesia due to long - term opium use was the predisposing factor for biliary cast formation in our case .
the efficacy of udca use in the treatment of persistently elevated liver enzymes following endoscopic removal intervention was reported previously .
the supposed mechanism by which udca acts in the treatment of bcs is dissolution of the cast in the small intrahepatic bile ducts , its hepatocytoprotective action and its effect on biliary acid secretion and immunomodulatory properties .
generally , this case brings up the effect of opium use on bcs , and with attention to the high prevalence rate of opium consumption in iran , this subject should be considered in these patients .
also , this case showed an effect of udca in the completion of treatment in bcs patients after cast removal .
it seems that biliary dyskinesia due to long - term opium use can be another predisposing factor for biliary cast formation , and that administration of udca after removal of the biliary cast is an effective treatment for bcs .
 biliary cast syndrome ( bcs ) is an uncommon complication which is mostly described in orthotopic liver transplantation . 
 however , bcs has also been reported rarely in non - liver transplant patients . 
 we describe a male long - term opium inhaler with bcs who underwent successful endoscopic cast removal by balloon enteroscopy - guided endoscopic retrograde cholangiopancreatography . a 52-year - old man , who was a known case of opium addiction , presented with the chief complaint of epigastric pain for 1 week prior to admission . 
 routine laboratory evaluation revealed cholestatic liver enzyme elevation . 
 a cholestatic pattern was seen in radiographic modalities . 
 endoscopic retrograde cholangiopancreatography showed a linear filling defect in the intra- and extrahepatic duct . 
 a long biliary cast was successfully removed using an extractor balloon . 
 after removal of the biliary cast the patient is receiving ursodeoxycholic acid and does not report any problem 4 months after treatment . 
 it seems that biliary dyskinesia due to long - term opium use can be a predisposing factor for biliary cast formation . 
Introduction
Case Report
Discussion
Conclusion