a biloma is a rare abnormal accumulation of intrahepatic or extrahepatic bile caused by traumatic or spontaneous rupture of the biliary tree1 , 2 ) .
it is most commonly caused by surgery , percutaneous transhepatic cholangiography ( ptc ) , percutaneous transhepatic biliary drainage ( ptbd ) , and abdominal trauma11 , 12 ) .
we report here a patient who complained of chills , fever and right upper quadrant abdominal discomfort .
a 78-year - old woman presented to the emergency room with her guardian , with acute fever and chills that developed the day before admission .
the patient was diagnosed with diabetes a few years previously , but this was not being treated .
on admission to the emergency room , the vital signs were as follows : blood pressure 70/40 mmhg , heart rate 118/min , respiratory rate 30/min and temperature 40.3. the patient appeared to be in acute distress ; she was lethargic , the sclerae were slightly yellow and the conjunctivae were pale .
the abdominal examination revealed slight tenderness in the right upper quadrant , but there was no palpable mass .
the laboratory studies were as follows : wbc 52,400/mm , hb 12.3 g / dl , platelets 90,000/mm , ast 153 iu / l ,
alt 135 iu / l , alkaline phosphatase 255 iu / l , total bilirubin 2.07 mg / dl , total protein 5.9 g / dl , albumin 3.0 g / dl , bun / cr 25/1.9 mg / dl , esr 25 mm / hr , and crp 20.4 mg / dl .
the serum electrolyte levels were normal except for an elevated potassium 7.0 mmol / l .
the abga data were as follows : ph 7.24 , pao271 mmhg , paco2 19 mmhg , hco3 15.1
the hepatitis viral marker tests , including hbsag , anti - hbs ab and anti - hcv , were all negative .
standard x - rays on the day of admission revealed lung infiltrates and no specific findings in the abdomen .
the abdominal computed tomography ( ct ) revealed a 9.58.5 cm subcapsular cyst with a clear boundary in the left hepatic lobe and enlargement of the intrahepatic bile duct around the cyst .
although the left hepatic lobe had some atrophic findings , there were no significant signs of an enlarged bile duct or abnormal gallbladder ( figure 1 ) . on the day of admission after the computed tomography ,
the abga revealed a ph 7.05 , pco2 64.8 mmhg , po2 73.3 mmhg , hco3
the patient was intubated and put on a respirator ; she was treated with antibiotics and vasopressors .
on the second day of admission , percutaneous drainage with an 8.5 fr pigtail catheter , under the guidance of an abdominal ultrasound was performed for diagnosis and treatment .
we removed 565 ml of fluid that was dark brown in color . for a diagnosis ,
biochemical tests were carried out on the bile , and these tests revealed a total bilirubin of 22.3 mg / dl and a direct bilirubin of 18.9 mg / dl .
the leukocyte count was 4,560/mm , and gram staining showed a gram negative bacillus . on the third day of admission ,
her vital signs were as follows : blood pressure 100/65 mmhg , heart rate 110/min and temperature 37.2. there was 200 ml of drained bile , the s - fibrinogen was 894 mg / dl ( 200 - 415 ) , the d - dimer was 9,040 ng / ml ( 0 - 255 ) , and the fdp was elevated up to 97 ug / ml ( normal range : 0 - 5 ) . on the eighth day of admission , the patient 's mental status improved , the vital signs were stable and voluntary respirations recovered .
on the thirteenth day of admission , pancreatico - biliary magnetic resonance imaging ( mri ) revealed a biloma that was connected to the intrahepatic bile duct ( figure 2 ) . on the sixteenth day of admission , the biloma that was connected to the intrahepatic duct and common bile duct
was noted again via a tubogram performed with a catheter . over time , the amount of bile drainage decreased . on the twentieth day of admission ,
64 ml of bile was drained , the leukocyte count was 340/mm and there were no signs of bacteria on culture or gram staining of the bile .
the follow - up ct showed that the size of the cyst had decreased to 5.44.6 cm and the density around the cyst had increased .
the amount of drainage did not decrease afterward , and the connection between the biloma and the bile duct was still noted . on the 25th day of admission ,
the drainage tube was blocked and follow - up of the bilirubin in blood and the size of the biloma were assessed . on the 29th day of admission ,
follow - up ct showed a marked decrease in the size of the cyst ( figure 3 ) , and the serum bilirubin was normal ( 0.22 mg / dl ) . on the 34th day of admission ,
the drainage tube was removed after evaluating the size and connection of the biloma via a catheter tubogram ( figure 4 ) .
they reported a case with extrahepatic bile leakage after trauma to the upper right quadrant of the abdomen ; the bile did not cause peritonitis , but it accumulated in an encapsulated form .
this concept was later applied to all cases with biliary tract lacerations caused by bile leaks that formed a capsule inside or outside of the liver1 ) .
the mechanism of encapsulation is explained by a large amount of bile leaking at a fast pace that causes biliary peritonitis , or a small amount of bile leaking at a slow pace that causes mild inflammation .
this leaked fluid is simultaneously trapped by the greater omentum and mesentery , where it becomes encapsulated2 ) .
although the amount of bile leaked may be minor , the occurrence of an inflammatory reaction contributes to the appearance of a growth in size of the biloma3 ) . as diagnostic techniques
continue to develop , more cases of biloma are being reported , but cases of spontaneous biloma and biloma accompanied by other diseases have rarely been reported to date . from 1979 to 1997 , 25 cases of spontaneous biloma were analyzed by fujiwara ; the underlying causes included choledocholithiasis ( 16 cases ) , cholangiocarcinoma ( 2 cases ) , acute cholecystitis ( 1 case ) , liver infarction ( 1 case ) , hepatic abscess ( 1 case ) , nephrotic syndrome ( 1 case ) , obstructive jaundice ( 1 case ) , sickle cell anemia ( 1 case ) , and tuberculosis ( 1 case)4 ) . in korea ,
there have been two cases of choledocholithiasis accompanied by a biloma , and they were treated by nonsurgical methods5 , 6 ) .
there is one report of a case of biloma after trauma7 ) , and 1 case of a biloma after endoscopic retrograde cholangiopancreatography ( ercp)8 ) .
one of the patients with a choledocholithiasis who suffered a nontraumatic intrahepatic rupture of the bile duct , had a surgical repair9 ) .
the causes of biloma include trauma to the liver , which is the most common cause , abdominal surgery , endoscopic surgery and percutaneous catheter drainage10 - 13 ) .
currently , the term biloma is generally used to describe intrahepatic or intraperitoneal focal biliary stasis14 ) .
the clinical symptoms of a biloma are nonspecific , and they can range from no symptoms to abdominal pain and distention , jaundice and fever ; leukocytosis can also be present15 ) . in the case
there were complaints of sudden chills , fever , upper abdominal discomfort and sensations of abdominal distention .
physical examinations showed tenderness in the upper right area of the abdomen , slight jaundice , hypotension , leukocytosis and fever over 40. the size and location of the biloma are determined by the cause of the rupture of the bile duct , the location and speed of bile leakage and the rate the bile is reabsorbed by the peritoneum2 ) .
fujiwara reported that the locations for bile leakage included the left hepatobiliary duct ( 8 cases ) , the gallbladder ( 5 cases ) , the right hepatobiliary tract ( 2 cases ) , the common bile duct ( 1 case ) , and unknown ( 9 cases ) ; the locations where the bilomas formed were the left lobe ( 11 cases ) , the right lobe ( 10 cases ) , and the upper abdomen ( 4 cases ) .
vazquez analyzed 21 cases of biloma and found that 16 cases occurred in the upper abdominal area near the liver and five in the left upper abdominal area4 ) .
the diagnosis of a biloma can be assisted by surgery , a history of trauma with abdominal ultrasonography , abdominal ct and mri .
a definite diagnosis can be made by a 99mtc - disida scan , percutaneous aspiration or by ercp1 , 2 ) .
abdominal ultrasonography shows a mass of low echogenicity with a clear border in the liver , and the follow up tests show changes in size16 ) .
the abdominal ct will give a more accurate view of the location of the biloma and show its relationship to the surrounding organs . a standard ct view
will show a low - density mass in the liver at a ct number lower than 20 .
ultrasonography and abdominal ct are sometimes unable to differentiate bilomas from seromas , lymphoceles and angiomas .
mris or hepatobiliary scans can be useful when continuous bile leakage is present , but they are not diagnostic when continuous leakage is not present7 , 17 ) . radiological image - guided aspiration tests , when testing for bilirubin , can be diagnostic18 ) .
when the aspirated substance is a clear yellow liquid , then microbiological tests must be done to rule out an infection .
ercp can be used to determine the location and severity of an active bile leak .
however , the presence of small biliary cysts or bilomas , located in the lower areas of the liver that can be hidden by gastrointestinal shadows , can be difficult to diagnosis19 ) .
we were able to identify a cystic lesion by abdominal ct ; a tubogram and magnetic resonance cholangiopancreatography ( mrcp ) were used to confirm a bile leak from the common bile duct .
the fluid obtained by the transabdominal ultrasonography assisted percutaneous cyst drainage containing 22.3 mg / dl of total bilirubin and 18.9 mg / dl of direct bilirubin ; these findings confirmed the diagnosis of a biloma .
treatment for bilomas that have a diameter of only a few centimeters is not always necessary ; these lesions can be watched . however , most bilomas require treatment . in the past , surgery was the main approach to treatment . currently , there are a much wider variety of options such as percutaneous catheter drainage , endoscopic sphincterotomy ( est ) , endoscopic nasobiliary drainage ( enbd ) and endoscopic drainage4 , 6 , 15 , 19 , 20 ) .
surgery is now performed only in cases with a persistent bile leak or for treatment of underlying disease . in this case , a spontaneous biloma with bacterial infection was found after the patient presented in septic shock .
after percutaneous drainage was performed , the amount of bile excreted did not decrease to lower than 64 ml per day . as there was no difficulty in flow through the common bile duct , the biochemical tests performed after removal of the drainage catheter showed no increase in total bilirubin or any increase in the size of the biloma , thus demonstrating a rare case of recovery .
 a " biloma " is a loculated collection of bile located outside of the biliary tree . 
 it can be caused by traumatic , iatrogenic or spontaneous rupture of the biliary tree . 
 prior reports have documented an association of biloma with abdominal trauma , surgery and other primary causes , but spontaneous bile leakage has rarely been reported . 
 a spontaneous infected biloma , without any underlying disease , is a very rare finding . 
 we recently diagnosed a spontaneous infected biloma by abdominal computed tomography and sonographically guided percutaneous aspiration . 
 the patient was successfully managed with percutaneous drainage and intravenous antibiotics . 
 we report here a case of infected biloma caused by spontaneous rupture of the intrahepatic duct , and review the relevant medical literature . 
INTRODUCTION
CASE REPORT
DISCUSSION