we present a unique case of a patient presenting with abdominal pain , obstructive jaundice and upper gastrointestinal haemorrhage resulting from a ruptured cystic artery pseudoaneurysm and concomitant cholecystojejunal fistula .
an 86-year - old male presented to the emergency department with a two - day history of colicky right upper quadrant ( ruq ) pain associated with bilious vomiting .
the patient was icteric with an associated pyrexia of 38  c , hypotension and tachycardia .
laboratory investigation revealed raised inflammatory markers with a white blood cell count of 24.0    10l and c - reactive protein of 95  mg / l ( normal range : < 7.5  mg / l ) .
liver function tests ( lfts ) were also deranged with an alkaline phosphatase of 450  /l ( 46111  /l ) , alanine transaminase of 142  /l ( < 45  /l ) and bilirubin of 60  mol / l ( < 20 
an ultrasound examination revealed a 5.7  cm calculus within the gallbladder surrounded by heterogeneous material ; a diagnosis of cholangitis was made .
the patient received fluid resuscitation with intravenous crystalloid and was commenced on broad - spectrum antibiotics .
subsequently , the patient passed a significant volume of malaena and fresh blood per rectum ( pr ) , associated with cardiovascular instability .
emergency oesophagogastroduodenoscopy ( ogd ) revealed fresh blood in the second part of the duodenum and a tubular clot adherent to the medial wall ( fig .
this was assumed to be originating from a duodenal ulcer and apparent haemostasis was achieved with injection of 15  ml of 1:10,000 adrenaline . however , the patient continued to pass malaena and pr blood following the procedure .
as such , a contrast - enhanced computed tomography ( ct ) scan was performed , demonstrating : ( 1 ) small bowel obstruction secondary to a gallstone impacted in the distal ileum ( fig .
2a ) ( 2 ) a thick - walled and inflamed gallbladder with evidence of recent intra - cholecystic bleeding ( fig .
2b ) , and ( 3 ) a 12  mm pseudoaneurysm emanating from the cystic artery ( fig . 2c and d ) .
in retrospect , the blood clot seen at ogd was likely to be haemobilia originating from the ampulla of vater .
the patient underwent emergency laparoscopy and the gallbladder was found to be necrotic and surrounded with dense adhesions .
division of these exposed a cholecysto - jejunal fistula containing clotted blood that was extending into the gallbladder .
evacuation of this clot revealed a 1  cm    2  cm pseudoaneurysm originating from the cystic artery , which began to bleed during dissection and the procedure was subsequently converted to an open laparotomy via a kocher incision .
a subtotal cholecystectomy was performed with oversewing of hartmanns pouch and the aneurysm was successfully resected from the gallbladder fossa .
the diseased jejunal loop was mobilised and the defect from the fistula was oversewn . despite detailed examination of the small bowel
the laparotomy wound was therefore closed under the assumption that the stone had passed spontaneously .
the patient was discharged following a fifteen - day inpatient stay and remained well at six - week outpatient follow - up .
cystic artery pseudoaneurysms remain a rare cause of upper gastrointestinal bleeding , with only twenty - two documented cases in the english literature .
they develop primarily as a consequence of adventitial damage and thrombosis of the vasa vasorum , resulting in damage to the muscular and elastic components of the media and intima with ensuing extravasation of arterial blood , progressive enlargement and eventual rupture , as governed by the law of laplace .
this can occur secondary to inflammatory conditions ( e.g. cholecystitis , pancreatitis ) , malignancy , biliary tract manipulation or trauma .
formation may be further accelerated by patient factors , such as atherosclerosis , hypertension , bleeding disorders and vasculitis .
cystic artery pseudoaneurysms tend to enlarge and erode into the gallbladder and adjacent biliary tree with approximately 45% bleeding into the biliary system ( haemobilia ) .
the clinical presentation is that of biliary colic ( 70% of cases ) , obstructive jaundice ( 60% ) and upper gastrointestinal bleeding ( 100% ) .
3240% of patients will present with all three symptoms  quincke 's triad . in the presented case ,
the patient had clinical and ct - confirmed cholecystitis in the presence of fistulating gallstones .
visceral inflammation adjacent to the cystic arterial wall is likely to have produced serosal ulceration and partial erosion of the elastic and muscular components of the arterial wall , thus leading to the formation and subsequent rupture of the pseudoaneurysm .
the pathological process of cholecystitis causing pseudoaneurysm formation is very rare , as inflammation usually results in cystic artery thrombosis .
therefore discuss that pseudoaneurysm formation is likely to be due to inflammation in the presence of a large gallstone eroding into the cystic artery , as may be the cause in our case .
haemorrhage from this pseudoaneurysm into the gallbladder thus resulted in haemobilia , with subsequent biliary colic and obstructive jaundice , as highlighted by lfts on admission .
cholecystoenteric fistulas alone are a rare complication of gallstone disease , with a reported incidence of 0.154.8% in patients with cholelithiasis undergoing biliary surgery , of which the majority are cholecystoduodenal ( 7180% ) .
one series examined over 15,000 laparoscopic cholecystectomies and highlighted the incidence of cholecystoenteric fistulae as being 0.2% ( n  =  34 ) with only five fistulae communicating with jejunum ( 14.7% ) 
fistulae arise when an obstructed cystic duct become repeatedly inflamed , resulting in adhesion of the gallbladder to an adjacent viscus , in this case the jejunum , with erosion of the intervening tissues and eventual fistulation .
this may be mediated by gallbladder wall necrosis from mechanical pressure due to an impacted gallstone .
most patients present with features of general gallstone disease but , less commonly , patients may present with cholangitis , severe upper gastrointestinal haemorrhage or gallstone ileus . in the reported case , the fistula resulted in gallstone ileus and rupture of the pseudoaneurysm produced haemobilia and upper gastrointestinal haemorrhage via the cholecystoenteric fistula .
describes a similar presentation in a patient presenting with lower gastrointestinal bleeding as the result of a ruptured cystic artery pseudoaneurysm with a concomitant cholecystocolonic fistula . to the best of the authors knowledge ,
this report describes the first case of a synchronous cystic artery pseudoaneurysm and cholecystojejunal fistula .
the initial investigation for a patient presenting in this manner is colour doppler ultrasonography , which will demonstrate a pseudoaneurysm as an anechoic lesion with colour flow through it .
this , however , has a low sensitivity and is limited when visualising sub - centimetre lesions , with one series demonstrating a detection rate of only 27% .
such was the case with our patient where ultrasound revealed a distended gallbladder with internal echogenicity but failed to identify the 12  mm pseudoaneurysm .
an ogd was subsequently performed revealing blood in the second part of the duodenum , which was likely to be arising from the ampulla of vater due to haemobilia .
upper gastrointestinal endoscopy is the best means of identifying haemobilia and , if seen , limits the focus to a hepato - biliary source of the bleeding
. however , endoscopy offers no means of intervention for haemobilia and , if intermittent , then this sign may be missed .
subsequent investigation is disputed in the literature between contrast - enhanced ct scanning and digital subtraction angiography .
argue that contrast - enhanced ct scanning and 3-dimensional ct angiography should replace arteriography as a non - invasive method of establishing a definitive diagnosis . however , many sources state that , although invasive , selective hepatic artery angiography is the gold standard for diagnosis , with a sensitivity of 80% and the ability to define aneurysms < 10  mm .
diagnostic angiography can then be combined with therapeutic arterial embolisation with foam , thrombin or micro - coils , either as definitive management or as a method of haemostasis prior to cholecystectomy and aneurysm repair .
there is a general consensus in the literature that emergency cholecystectomy is the treatment of choice in haemobilia due to cholecystitis , gallstones or resectable neoplasms , or if embolisation fails . where possible , however , a two - stage approach should be adopted to managing cystic artery pseudoaneurysms : with embolisation of the bleeding pseudoaneurysm first in order to stabilise the patient followed subsequently by cholecystectomy . in our case , it was not possible to perform diagnostic angiography and so contrast - enhanced ct angiography was the investigation of choice .
this not only clearly demonstrated the cystic artery pseudoaneurysm but also revealed gallstone ileus and the associated fistula . if available , arteriography and embolisation would have been appropriate to stabilise the patient but these pathologies identified on ct - imaging required urgent operative intervention and , as such , emergency laparoscopy with subsequent conversion to laparotomy was performed .
in conclusion , we present a unique case of a ruptured cystic artery pseudoaneurysm causing haemobilia and upper gastrointestinal bleeding through a concurrent cholecystojejunal fistula .
we have demonstrated the success of surgical management alone in the treatment of such a case but accept that , when available and clinically indicated , a combined therapeutic approach with angiography should be considered in the first instance .
 introduction cystic artery pseudoaneurysms and cholecystoenteric fistulae represent two rare complications of gallstone disease.presentation of case an 86 year old male presented to the emergency department with obstructive jaundice , ruq pain and subsequent upper gastrointestinal bleeding . 
 upper gi endoscopy revealed bleeding from the medial wall of the second part of the duodenum and a contrast - enhanced computed tomography scan revealed a cystic artery pseudoaneurysm , concurrent cholecystojejunal fistula and gallstone ileus . 
 this patient was successfully managed surgically with open subtotal cholecystectomy , pseudoaneurysm resection and fistula repair.discussion to date there are very few cases describing haemobilia resulting from a bleeding cystic artery pseudoaneurysm . 
 this report is the first to describe upper gastrointestinal bleeding as a consequence of two synchronous rare pathologies : a ruptured cystic artery pseudoaneurysm causing haemobilia and bleeding through a concurrent cholecystojejunal fistula.conclusion through this case , we stress the importance of accurate and early diagnosis through ultra- sonography , endoscopy , and contrast - enhanced ct imaging and emphasise that haemobilia should be included in the differential diagnosis of anyone presenting with upper gastrointestinal bleeding . 
 we have demonstrated the success of surgical management alone in the treatment of such a case , but accept that consideration of combined therapeutic approach with angiography be given in the first instance , when available and clinically indicated . 