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Classical Case Studies

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Neil Mackie, Sudheer Vinnamala, Matthew Lewis A case of Mirizzi's syndrome and cholecystocolonic fistula
Neil Mackie, Sudheer Vinnamala, Matthew Lewis, 11 March 2013

Summary

We describe the case of a 72-year-old lady presenting with abdominal pain and fever secondary to gall stone cholecystitis, confirmed on ultrasound scan.  She was treated conservatively with antibiotics and reviewed in clinic to be assessed for interval cholecystectomy. 

Cholecystectomy was not performed because she was too high risk for surgery due to COPD. She subsequently developed diarrhoea, weight loss and abdominal pain as a result of diagnosed Mirizzi's syndrome which was later complicated by cholecystocolonic fistula.

A large irremovable stone was identified at ERCP. Eventually, the stone migrated into the gut and was passed spontaneously per rectum.

Case Report

History

A 72-year-old lady presented via the Emergency Department complaining of a long history of right-sided abdominal pain and recent fevers in late 2005.

No past history of abdominal pathology was noted, although the patient reported recurrent chest infections and a long smoking history.

The only abnormality on serum analysis was a significantly raised CRP.

She received antibiotics and was discharged with follow-up at surgical clinic and outpatient abdominal ultrasound.

At follow up, the patient continued to complain of intermittent right upper quadrant, iliac fossa and flank pain with reduced appetite but no further pyrexia. Ultrasound demonstrated multiple gallbladder stones.

Outpatient OGD and colonoscopy were arranged to investigate her symptoms which were not considered to be typical of gallbladder disease.

Examination

Examination revealed only tenderness in the right flank and iliac fossa. The patient was not jaundiced. No abdominal masses were noted. Investigation and colonoscopy revealed only the presence of a small (< 5cm) hiatus hernia.

CT abdomen was arranged to investigate other causes of the ongoing abdominal pain and recent weight loss. CT confirmed the presence of hiatus hernia and demonstrated multiple stones in a porcelain gallbladder.

 

 

Fig 1:  CT showing presence of multiple stones and porcelain gall bladder.

 

Management

The patient was subsequently assessed for cholecystectomy, but was not felt to be suitable due to poor respiratory reserve secondary to significant smoking history. Her abdominal pain was well controlled with oral analgesia, and the patient reported no further weight loss.

Outcome:

In early 2009, the patient re-presented to the general surgeons complaining of painless jaundice and weight loss. On examination a palpable liver was noted. Liver function tests were deranged with predominant cholestasis. She was investigated with CT abdomen, which noted the presence of porcelain gallbladder and biliary dilatation. The patient went on to have ERCP, which found a single biliary stenosis in the upper third of the CBD. The proximal bile ducts were dilated and a plastic stent was inserted for biliary drainage. No evidence of malignancy was found on CT or from brushings taken at ERCP. She was also noted to have persistent diarrhoea at this point.

 

 

Fig 2:  AXR with visible gallbladder.

 

 

Fig 3:  CT demonstrating presence of heavily calcified GB.

 

Four months later, the patient was admitted to the gastroenterology department complaining of nausea, vomiting and diarrhoea. Serum screen for malabsorption was normal. She underwent further OGD and colonoscopy, followed by repeat abdominal CT as the sigmoid colon was fixed at colonoscopy.

The scan showed a likely stone at the junction of the cystic and hepatic ducts. Also noted were: migration of the biliary stent into the duodenum, pneumobilia and inflammation and thickening around the colonic mucosa at the hepatic flexure. The patient also had a persistently raised ALP since presentation to the surgeons, although her jaundice had resolved. The patientís diarrhoea was diagnosed as bile salt malabsorption and she was commenced on cholestyramine.

 

Fig 4:  CT demonstrating pneumobilia and colonic wall thickening.

 

The patient underwent repeat ERCP, attempting to remove the gallstone noted on CT. ERCP confirmed a single impacted stone in the gallbladder neck, occluding the common hepatic duct (Mirizzi's syndrome). The size and position of stone prevented its removal during the ERCP, and a fresh biliary stent was inserted. There was no jaundice at this stage.

The patient was referred to surgeons to assess her suitability for roux-en-y resection. The patient declined surgical treatment owing to the high risk of complication and potential mortality. She reported no further weight loss, and management continued with biliary stenting to prevent jaundice and cholestyramine for her bile salt diarrhoea.

 

Fig 5:  ERCP showing outline of impacted stone compressing hepatic duct.

During a stent change in late 2010, the upper CBD could no longer be accessed, and contrast leak in to the large bowel was noted. The patient was admitted for further investigation with abdominal CT, which confirmed the presence of contrast in the transverse colon, but no evidence of intraperitoneal leak. A replacement biliary stent was successfully placed on repeat ERCP. The patient subsequently developed two acute episodes of cholangitis, which resolved completely with antibiotics.

 

Fig 6:  ERCP image demonstrating contrast leak.

 

Fig 7:  CT demonstrating leak of ERCP contrast into transverse colon.

 

In early 2012, the patient was investigated by the respiratory department for incidental pulmonary nodules. Serial CTs showed resolution of her pulmonary nodules, which were diagnosed as inflammatory and benign in nature. The patient was then admitted a month later for treatment of pneumonia, during which time she was also investigated with CT abdomen as she reported continued weight loss and abdominal pain. The CT demonstrated migration of the previously impacted gallstone into the sigmoid colon and the patient was noted to pass the gallstone in her stool during admission. She kept the stone in order to present it to the gastroenterology department several months later.

 

 

Fig 8 and Fig 9: Serial CT abdomens showing migration of stone from GB to sigmoid colon (taken 1/12 apart).

 

 

Fig 10: The gallstone, taken several months after passage.

 

Mirizzi syndrome and cholecystocolonic fistula

Mirizzi syndrome describes the extrinsic compression of part or whole of the biliary tree by an impacted gallstone within the gallbladder neck. When untreated, this can lead to necrosis of the duct wall and recurrent cholangitis. Subsequent formation of fistulae has been reported, most commonly between the gallbladder and an area of the biliary tree. Mirizzi syndrome can be classified (grades I to IV) by the presence and size of these cholecystobiliary fistula [1].

If the impacted gallstone is not removed, the patient is at risk of more fistulae forming between the gallbladder and surrounding viscera, particularly between the gallbladder and bowel. Gallstone disease can be identified as the underlying cause of enterobiliary fistulation in 90% of cases [2] [3].

Enterobiliary (EB) fistulae are not exclusively secondary to gallstone erosion and have been demonstrated in inflammatory bowel disease, malignancy, peptic ulcer disease and following trauma. Enterobiliary fistulae are most commonly cholecystoduodenal (70% of EB fistulae), with only 10-20% of EB fistulae being cholecystocolonic [2].  Fistula formation has also been associated with increasing age and is more common among females [3] [4]. Whether or not this is simply because the presence of gallstones is more prevalent in older females is unclear.

Diarrhoea, malabsorption, nausea, abdominal pain and weight loss are common in patients with cholecystoenteric fistula. This is suspected to be due interruption of the normal entero hepatic bile circulation, leading to bile salt malabsorption and which usually responds well to cholestyramine. The presence of bile salts within the colon also produces osmotic diarrhoea, contributing to symptoms [4][6]. This is consistent with the clinical features the patient developed in this case. Definitive treatment is to remove the impacted gallstone by either laparoscopy or laparotomy with resection of the fistulated section of bowel [2] [3] [4].  Small stones may be removed at ERCP.

In this case, the impacted stone was too large to remove by ERCP without causing unacceptable tissue damage and the patient was not suitable for surgical management due to her high anaesthetic risk. Cholecystectomy was not performed at an early stage due to this risk and the initial resolution of symptoms of gallstone disease following antibiotics alone.

The earliest features of cholecystoenteric fistula were seen in 2009, when the patient presented with diarrhoea, and CT at the time reported pneumobilia with inflammation of the colonic mucosa. At this point, the increased risk associated with performing the required roux-en-y resection ruled out surgery as a viable treatment option.

The goal of the treatment she then received was symptom control: stent insertion to bypass the obstructing gallstone and relieve jaundice; oral bile acid sequestrants (cholestyramine) to control her persistent diarrhoea and nutritional support through oral nutritional supplements to prevent further weight loss.

She has not had any further episodes of cholangitis since passage of the gall stone and her diarrhoea is now well controlled with cholestyramine use.

 

References

  1.   Chatzoulis G, Kaltsas A et al.  Mirizzi syndrome type IV associated with cholecystocolonic fistula: a very rare condition; 27 May 2007.

  2. Angel MRR, Clarke L. Cholecystoclonic fistula as a complication of cholelithiasis: a rare case; Internet Scientific Publications.

  3. Correia M, Amonkar D. Cholecystocolic Fistula: a Diagnostic Enigma; Saudi J Gastroent 2009 15(1) 42-44.

  4. Chen C. Cholecysto-colonic fistula: a case report; New York Med J.

  5. Hession PR, Rawlinson J. The clinical and radiological features of cholecystocolic fistula; British J Radiology 1996 Sept 69(825) 804-9.

  6.   Elsas LJ, Gilat T. Cholecystocolonic fistula with malabsorption. Annals of Internal Medicine; Sept 1965 63(3) 481-486.
 
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 25 July 2014

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