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Case Report

Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication

[version 1; peer review: 3 approved]
PUBLISHED 27 Sep 2017
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Abstract

One of the most unusual complications in cholethiasis is spontaneous cholecystocutaneous fistula, which has only been reported a few times in the literature.  We report the case of a 76 year old man who presented with a right hypochondrium subcutaneous abscess, with pain evoked through palpation.  No comorbidity in the patient’s medical history were noted.   Confirmation of cholecystocutaneous fistula was made using the proper diagnostic process, which is computed tomography with contrast media, followed by hepatobiliary MRI. This confirmed the presence of a fistulous pathway between the gallbladder and the skin.  The patient underwent cholecystectomy surgery and open laparotomy with en block aponeurotic muscle, skin and fistula orifice excision.

Keywords

Cholecystocutaneous, Fistula, cholethiasis, abdominal fistula, gallbladder stones, gallbladder cancer, Bile Ducts, Laparotomy

Introduction

Spontaneous cholecystocutaneous abscess or fistula is an extremely uncommon complication of gallbladder disease. Less than 100 cases have been described in the literature. The first descriptions of cholecystocutaneous fistula was made by Thelisus in 1670. Later Courvoisier reported 169 cases of biliary fistula in the 19th century1. The natural history of the disease has changed from suppurative cholecystitis with spontaneous rupture to operative external drainage of an abscess2. Early and effective medical and surgical management of biliary tract disease can prevent this rare condition. Stone obstruction of the biliary tree plays a crucial role in the pathophysiology of the development of this condition; intra-gallbladder pressure can increase dramatically due to the obstruction of the cystic duct. The unresolved obstruction of the bile outflow compromise gallbladder wall blood circulation, as well as lymphatic drainage, resulting in necrosis of the gallbladder wall with the fistula formation. Once pierced, the gallbladder may drain into the peritoneal cavity, causing peritoneal localized abscess or the abscess can lead into an external fistula due to its adherence to the abdominal wall13.

Case report

A 76 year old man was admitted to our University Hospital “Ospedale Vittorio Emenuele” and seen in our surgical department. He presented with a 3 cm tumefaction of the right hypochondrium, surrounded by an erythematous skin area, with small secretion of a yellowing-green material, attributable to a bile leaking (Figure 1). The patient’s medical history was clear from previous medical disease and surgery; he only referred to previous upper right quadrant pain and nonspecific dyspeptic disorder.

77cf9913-732b-4452-b41a-71dbda129c92_figure1.gif

Figure 1. Cholecystocutaneous fistula, macroscopic appearance.

An abdominal ultrasound examination revealed the presence of a lesion in the aponeurotic muscle wall, but the possible underlying pathology was unknown. No other signs of pathology were observed. Routine blood work was normal.

Abdominal computed tomography scan with contrast media showed the gallbladder walls had diffuse thickened and blurred edges, and the right and transverse abdominal muscles were almost covered and embedded with minute hypo-dense ailments compatible with relapsing phlogistic processes (Figure 2). Hepatobiliary MRI detected that the gallbladder had anteriorly shifted and adhered to the right abdominal muscles. The side wall showed a break through with consensual purulent collection, which extruded through the thick abdominal wall (Figure 3). Eventually, several different-sized stones were revealed inside the cholecyst. Consequently, a diagnosis of spontaneous cholecystocutaneous fistula was made.

77cf9913-732b-4452-b41a-71dbda129c92_figure2.gif

Figure 2. Abdominal computed tomography images.

Left image, * indicates the gallbladder; right image, the arrows indicates the fistula.

77cf9913-732b-4452-b41a-71dbda129c92_figure3.gif

Figure 3. Hepatobiliary MRI.

The white arrows show the fistula. From left to right: coronal and sagittal fistula view.

The patient underwent cholecystectomy surgery 10 days after diagnosis, with an open laparotomy with en block aponeurotic muscle, skin and fistula orifice excision. In order to have a good abdominal wall reconstruction, a properly shaped Prolene Prosthesis was placed using fibrin glue47. The patient received broad-spectrum antibiotics after surgery (Piperacillin/Tazobactam 4.5 g, 3 times a day for 5 days via IV).

Considering the patient’s good general condition and good post-operative course, discharge was on the seventh day post-surgery. Surgical wound re-dressing was made one week after discharge in our facility, where the surgical stitches were removed. Scar appearance was good and without concern. No other dressing was needed and the scar was covered with a bandage. The first follow-up was scheduled 15 days after discharge, second one 60 days from discharge. For both follow-ups, routine blood work and surgical scar checking were performed, the results of which were normal and the scar was healing normally.

A histological examination confirmed the diagnosis of chronic cholecystitis with gallstones and cholecystocutaneous fistula.

Discussion

Thanks to the progress made with medical imaging and surgical techniques, biliary fistula is today a very rare pathology811. Fistulas often represent the result of post-surgical12 or post-traumatic15 complications that generally involve the duodenum (77%) and colon (15%)16.

Spontaneous cholecystocutaneous fistula represents a truly exceptional event, as confirmed by the analysis of the literature, which revealed only 28 cases published over the last 10 years (Table 1). This disease mainly affects female subjects over the age of 60. Etiology is generally due to an acute inflammatory process as a consequence of a cholecystitis or chronic gallstones disease1720, although there are described cases of spontaneous cholecystocutaneous fistula in the absence of gallstones21. Rarely does cholecystocutaneous fistula evolve into a neoplastic process. Instead, fistula can be a sign of gallbladder cancer19,20. According to Sibakoti, polyarteritis nodosa with gallbladder vasculitis and prolonged use of high dose steroids can be considered predisposing factors21. Fistula primum movens is by cystic duct obstruction, which increases the pressure within the gallbladder, with wall distension and impaired vascularization, resulting in the formation of focal necrosis of the wall with perforation evolution and abscess formation to the surrounding area that will rupture in to the continuous structures. In the present case, the abscess drained through the abdominal wall and the fistulose pathway originated from the bottom of the gallbladder. This area is the most distant from the cystic artery and physiologically the least vascularized and therefore more susceptible to ischemia17.

Table 1. Publications within the last 10 years concerning cholecystocutaneous fistula.

Author(s)Year
published
Number
of cases
CountryAgeGenderTreatment
technique
Maynard et al.3520161United Kingdom68FOpen
Jayasinghe et al.3620161United Kingdom>70FOpen
Guardado-B et al.3720151Mexico30FOpen
Álvarez et al.3820141Argentina79FOpen
Dixon et al.3920141United Kingdom94FOpen
Pripotnev and
Petrakos40
20141Canada85FOpen
Kim et al.4120131AustraliaOpen
Jayant et al.4220131India42FOpen
Sodhi et al.4320121India66FOpen
Kapoor et al.4420131India45MOpen
Ozdemir et al.4520122India45 & 65MOpen
Ugalde Serrano et al.4620121Spain83MOpen
Andersen and
Friis-Andersen24,47
20121Denmark89FOpen
Ioamidis et al.4820121Greece71MOpen
Cheng et al.4920111ChinaOpen
Gordon5020111U.S.A.83FOpen
Sayed et al.5120101United Kingdom85FOpen
Pezzilli et al.5220101Italy90FOpen
Metsemakers et al.5320101Belgium69MOpen
Tallón Aguilar et al.5420101Spain83FOpen
Kahn et al.5520101Ireland76MOpen
Hawari et al.5620101United Kingdom84MOpen
Murphy et al.5720081United Kingdom80MOpen
Ijaz et al.5820081United Kingdom80FOpen
Chatterjee et al.5920071India45FOpen
Malik et al.6020071United Kingdom76FLaparoscopic

The external fistular orifice is usually on the right upper quadrant, but other locations have been described, including the left hypocondrium, umbilical scar, right lumbar, and right iliac fossa, and rarely the right gluteus and breast region19,24,47.

The diagnostic process always begins with upper abdomen ultrasound and ends with hepatobiliary MRI to visualize the biliary tree. Considering that 11% of cholecystitis have concomitant presence of gallstones in the main bile duct, it is advisable to perform endoscopic retrograde cholangiopancreatography (ERCP)229. In our case CT with CM and hepatobiliary MRI confirmed the fistula presence and it was not necessary to execute the ERCP before surgery. Although an intraoperative cholangiogram was performed to check that the bile ducts were clear from gallstones. Cholecystocutaneous fistula has always been treated by two different strategies. The first includes a two-step approach: percutaneous drainage and antibiotic therapy, and subsequently cholecystectomy. The second directly involves laparotomy cholecystectomy execution with en block aponeurotic muscles, as well as skin and fistula orifice excision.

The second strategy is the most commonly used since the two-step approach treatment is reserved for patients with sepsis and poor general condition12,15,29.

In 1998, Kumar described the first case of gynecological fistula treated with laparoscopic technique, proposing to the scientific community the feasibility of this innovative approach27,30.

Conclusion

Rarity of this pathology confirms the great quality of progress made by early diagnostic techniques and medical treatment to prevent complication of cholethiasis. Although cholecystocutaneous spontaneous fistula is not common, it can lead to a serious condition. If not quickly treated, it can rapidly evolve into a generalized septic state with severe impairment prognosis. In our case, the patient was in good health arguably because the fistula was draining the most of the abscess outside the body and not in the peritoneum space. Surgical treatment was, however, essential to restore the physiologic bile flow and adequate broad-spectrum antibiotic prophylaxis lowed the risk of post-operative infections. Although laparoscopic approaches have been described since 1998, this pathology is, in most cases, continuing to be treated with open technique, most likely because it is easier and with fewer risks of post-surgical complications3134.

Consent

Written informed consent was obtained from the patient for the publication of the patient’s clinical details and related images.

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Rinzivillo NMA, Danna R, Leanza V et al. Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication [version 1; peer review: 3 approved]. F1000Research 2017, 6:1768 (https://doi.org/10.12688/f1000research.12235.1)
NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article.
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ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested
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Not approvedFundamental flaws in the paper seriously undermine the findings and conclusions
Version 1
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PUBLISHED 27 Sep 2017
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Reviewer Report 24 Nov 2017
Giuseppe Navarra, Surgical Oncology Unit, Department of Human Pathology of Adult and Evolutive Age, University Hospital of Messina, Messina, Italy 
Approved
VIEWS 2
The article Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication covers a subject that nowadays is quite uncommon. It goes through the background of the case’s history, it provides details of diagnostic tests, treatment given and outcomes reached. It could be useful for other ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Navarra G. Reviewer Report For: Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication [version 1; peer review: 3 approved]. F1000Research 2017, 6:1768 (https://doi.org/10.5256/f1000research.13246.r28008)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
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5
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Reviewer Report 21 Nov 2017
Antonino Agrusa, Department of General Surgery, Urgency and Organ Transplantation, University of Palermo, Palermo, Italy 
Approved
VIEWS 5
My compliments to the authors for the interesting and rare case report. I approve it for pubblication in F1000 Research.

Please give more explanation for the 10 days delay between patient admission and operative surgical management. On the basis of rare case ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Agrusa A. Reviewer Report For: Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication [version 1; peer review: 3 approved]. F1000Research 2017, 6:1768 (https://doi.org/10.5256/f1000research.13246.r28162)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
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10
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Reviewer Report 20 Nov 2017
Matteo Novello, Colorectal Surgery Department, Cleveland Clinic, Cleveland, OH, USA 
Salomone Di Saverio, General Surgery Department, Ospedale Maggiore, Bologna, Italy 
Approved
VIEWS 10
The authors should clarify the reasons for the 10 days delay in the operative management of their patient.

More insight should be provided on the choice of aggressive operative treatment as compared to a percutaneous drainage. Perhaps ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Novello M and Di Saverio S. Reviewer Report For: Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication [version 1; peer review: 3 approved]. F1000Research 2017, 6:1768 (https://doi.org/10.5256/f1000research.13246.r27856)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.

Comments on this article Comments (0)

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Alongside their report, reviewers assign a status to the article:
Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions
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