Cholecystocutaneous Fistula 

  • Author: Cherry Ee Peck Koh, FRACS, MBBS, MS; Chief Editor: John Geibel, MD, DSc, MA   more...
 
Updated: Aug 5, 2011
 

Background

Biliary fistulae can be divided into internal and external biliary fistulae. External biliary fistulae, in turn, can be further subdivided based on etiology into spontaneous, therapeutic, traumatic, and iatrogenic fistulae. Spontaneous external cholecystocutaneous fistula is a rare surgical complication of neglected calculous biliary disease that has become even increasingly rarer because of prompt diagnosis and expedient surgical intervention for gallstones.

Thilesus first described cholecystocutaneous fistula in 1670.[1] Prior to 1900, 3 large series were published in quick succession, including a report by Courvoisier in 1890 (169 of 499 cases of gallbladder perforation), 1 by Naunyn in 1896 (184 cases), and another by Bonnet in 1897 (122 cases).[2, 3, 4, 5]

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History of the Procedure

Since the advent of surgical treatment of gallbladder calculous disease, the incidence of cholecystocutaneous fistula has reduced dramatically; from 1890-1949, only 37 cases were identified in the published literature.[5] A literature review of cases published in the last 50 years reveals fewer than 50 cases (see Table). The declining incidence has been attributed to prompt diagnosis, availability of antibiotics, and early surgical intervention for cholecystitis and empyema (see Pathophysiology). The decreasing incidence is further confirmed by the availability of large series published prior to the 20th century, in contrast to more recent literature, which consists mainly of individual case reports.

Table. Case Reports of Cholecystocutaneous Fistula in the Past 50 Years From 1961 to 2011 (Open Table in a new window)

Author(s)Year PublishedNumber of CasesCountry of Origin
Gordon et al[6] 20111United States of America
Sayed et al[7] 20101United Kingdom
Pezzilli et al[8] 20101Italy
Metsemakers et al[9] 20101Belgium
Tallon Aquilar et al[10] 20101Spain
Hawari et al[11] 20101United Kingdom
Gandhi et al[12] 20091New Zealand
Murphy et al[13] 20081United Kingdom
Ijaz et al[14] 20081United Kingdom
Chatterjee et al[15] 20071India
Malik et al[16] 20071United Kingdom
Nagral et al[17] 20071India
Marwah et al[18] 20071India
Shrestha et al[19] 20061United Kingdom
Cruz et al[20] 20061Brazil
Salvador-Izquierdo et al[21] 20061Spain
Yuceyar et al[22] 20051Turkey
Khan et al[23] 20051Saudi Arabia
Dutriaux et al[24] 20051France
Gossage et al[25] 20041United Kingdom
Vasanth et al[26] 20041United States of America
Mathonnet et al[27] 20021France
Chang et al[28] 20021Taiwan
Flora et al[29] 20011United Kingdom
Ramos Rincon et al[30] 20011Spain
Nicholson et al[31] 19991United States of America
Avital et al[32] 19981Israel
Kumar[33] 19981United States of America
Andley et al[34] 19961India
Birch et al[35] 19911United Kingdom
Carragher et al[36] 19901United Kingdom
Rosario et al[37] 19901United States of America
Sevonius et al[38] 19881Sweden
Gibson et al[39] 19871United Kingdom
Bilanovic et al[40] 19871Croatia
Tuna et al[41] 19861United States of America
Hakaim et al[42] 19861United States of America
Rye et al[43] 19851Denmark
Kulicki et al[44] 19841Poland
Davies et al[45] 19841United Kingdom
Abril et al[46] 19841United States of America
Nayman[47] 19831Australia
Ulreich et al[48] 19831United States of America
Hoffman et al[49] 19821United States of America
Fitchett et al[50] 19701United States of America
Callen[51] 19791United States of America
Orr[52] 19791Australia
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Problem

A fistula is an epithelium-lined tract between 2 epithelium-lined surfaces. Thus, a cholecystocutaneous fistula is an abnormal epithelial tract that allows communication between the gallbladder and the skin. This communication can be either spontaneous or deliberate as in the case of a therapeutic percutaneous cholecystostomy used to treat cholecystitis or empyema of the gallbladder, which is generally reserved for patients unfit for surgical intervention, while the former is often a complication of neglected gallstone disease.

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Epidemiology

Frequency

Spontaneous cholecystocutaneous fistula is rare. No data exist for the incidence of this condition in the United States. In a retrospective review in Greece, of 210 cases of external biliary fistulae over a 22-year period, only 1 was due to spontaneous cholecystocutaneous fistula.[53]

To emphasize the rarity of this condition, from 1890-1949, only 37 published cases were found worldwide. After a careful search of indexed and nonindexed journals, fewer than 50 cases have been reported worldwide (see Table above). A closer look at the Table also reveals that just over half the cases have been reported in the past 10 years, which reflects the increased number of avenues for publication of case reports and recognition that the condition is rare.

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Etiology

This condition is invariably a complication of neglected gallstone disease, although isolated case reports have described spontaneous cholecystocutaneous fistula due to carcinoma of the gallbladder and acalculous cholecystitis.[54]Carcinoma of the gallbladder can cause cystic duct obstruction, which leads to inflammation in a manner similar to that of gallstones.[55]

In addition, retained gallstones following laparoscopic cholecystectomy may cause biliary fistula or abdominal wall sinuses. This occurs because gallstones can harbor bacteria, which may form a localized abscess with fistula or sinus in an attempt to discharge the foreign body.[56, 57] Although this is relatively uncommon despite a relatively common occurrence of stone spillage, some authors recommend the liberal use of retrieval bags during surgery to avoid stone spillage and subsequent complications of retained stones.[58]

Salmonella typhi, which has a predilection for the gallbladder, can cause chronic cholecystitis and may predispose the patient to spontaneous cholecystocutaneous fistula.[59] Polyarteritis nodosa with gallbladder vasculitis and steroid use causing immunosuppression also may be associated with the condition.[59]

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Pathophysiology

The cystic duct or gallbladder is almost always obstructed in patients with spontaneous cholecystocutaneous fistula. In the presence of obstruction, the gallbladder distends and the pressure within rises, impairing the vascular supply. The obstruction and impaired blood supply result in inflammation and may cause focal areas of necrosis. This inflammatory process is typically insidious and recurrent. Surrounding structures wall off the focal area of necrosis. Perforation of the gallbladder may occur, causing a localized cholecystic abscess. In an attempt to discharge this abscess, a fistula may thus form between the gallbladder and the duodenum, colon, or abdominal wall.

In spontaneous cholecystocutaneous fistula, the abscess is walled off by the abdominal wall and progressively penetrates it. The fistula usually occurs via the fundus of the gallbladder, as this is the farthest from the cystic artery and most likely to be affected in inflammation-caused ischemia. The cholecystic abscess may initially cause a tender area in the abdominal wall and spontaneously rupture, forming a fistula with drainage onto the skin.

Because of the anatomy and position of the gallbladder, the gallbladder much more commonly adheres to neighboring viscera, such as the duodenum and colon, forming cholecystoduodenal fistula that predisposes to gallstone ileus or cholecystocolonic fistula. Similar to cholecystocutaneous fistula, the incidence of cholecystoduodenal fistula is also declining because of expedient surgical intervention.

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Presentation

Epidemiology

Patients tend to be elderly, although cholecystocutaneous fistulae have been reported as early as the third decade of life. Younger patients are likely to have neglected their symptoms for a period of time or have neuropathy that causes altered sensation.[60] Women are affected more than men, reflecting the higher incidence of cholelithiasis and cholecystitis among women.

History

Most patients have a history suggestive of biliary disease; however, these symptoms are unlikely to be severe, as an acute attack would have precipitated early presentation for surgical intervention. Depending on the stage of progression, patients may present with empyema necessitatis or a discharging sinus.[61] Empyema necessitatis simply refers to a cholecystic abscess prior to rupture. The patient may report systemic symptoms, such as fevers, sweats, and anorexia associated with the infection, or a tender lump at the site of impending perforation. Patients in whom fistulae have discharged may report loss of bilious fluid or small stones via the external opening. The fistula itself is usually painless.

Examination

The patient may be febrile and diaphoretic because of the infection. Prior to rupture, a raised, erythematous, tender, hot area of affected skin may be observed (see the image below). The surrounding skin is often cellulitic, frequently leading to an initial diagnosis of abscess. An associated lump under the skin may be observed if the gallbladder has herniated through the overlying tissue or if an associated malignancy is present.

A 90-year-old man referred with abdominal wall absA 90-year-old man referred with abdominal wall abscess in the right upper quadrant.

The external opening is usually in the right upper quadrant, although external openings in the periumbilical area, the lumbar area, and even the gluteal area have been described. The discharge varies depending on whether an obstruction is present. Discharge may be purulent in the presence of empyema, mucoid in the presence of a mucocele because of obstruction, or bilious in the absence of obstruction. Small stones within the discharge often confirm the diagnosis.

Differential diagnosis

  • Infected epidermal inclusion cyst
  • Discharging tuberculoma
  • Pyogenic granuloma
  • Chronic osteomyelitis of ribs with sequestrum
  • Metastatic carcinoma
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Relevant Anatomy

See Pathophysiology.

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Contraindications

See Surgical therapy.

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Contributor Information and Disclosures
Author

Cherry Ee Peck Koh, FRACS, MBBS, MS  Surgical Registrar, Department of General Surgery, Alfred Hospital, Bayside Health

Cherry Ee Peck Koh, FRACS, MBBS, MS is a member of the following medical societies: Royal Australasian College of Surgeons

Disclosure: Nothing to disclose.

Coauthor(s)

David Merenstein, MBBS, FRACS  Consulting Staff, General and Endocrine Surgery, Department of Surgery, Monash Medical Centre, Faculty of Medicine, Nursing and Health Services; Consulting Staff, Sandringham and District Hospital, William Angliss Hospital and West Gippsland Hospital

David Merenstein, MBBS, FRACS is a member of the following medical societies: Royal Australasian College of Surgeons

Disclosure: Nothing to disclose.

Simon Roger Berry, MBBS  General, UGI, and HPB Surgeon, Surgical Consulting Group, Cabrini Hospital, Australia

Simon Roger Berry, MBBS is a member of the following medical societies: Australian Medical Association, Australian Medical Association, International Hepato-Pancreato-Biliary Association, and Royal Australasian College of Surgeons

Disclosure: Nothing to disclose.

Specialty Editor Board

Brian James Daley, MD, MBA, FACS, FCCP, CNSC  Professor, Associate Program Director, Department of Surgery, Division of Trauma and Critical Care, University of Tennessee Health Science Center College of Medicine

Brian James Daley, MD, MBA, FACS, FCCP, CNSC is a member of the following medical societies: American Association for the Surgery of Trauma, American College of Chest Physicians, American College of Surgeons, American Medical Association, Association for Academic Surgery, Association for Surgical Education, Eastern Association for the Surgery of Trauma, Shock Society, Society of Critical Care Medicine, Southeastern Surgical Congress, and Tennessee Medical Association

Disclosure: Nothing to disclose.

Francisco Talavera, PharmD, PhD  Adjunct Assistant Professor, University of Nebraska Medical Center College of Pharmacy; Editor-in-Chief, Medscape Drug Reference

Disclosure: Medscape Salary Employment

David L Morris, MD, PhD, FRACS  Professor, Department of Surgery, St George Hospital, University of New South Wales, Australia

David L Morris, MD, PhD, FRACS is a member of the following medical societies: British Society of Gastroenterology

Disclosure: RFA Medical None Director; MRC Biotec None Director

Paolo Zamboni, MD  Professor of Surgery, Chief of Day Surgery Unit, Chair of Vascular Diseases Center, University of Ferrara, Italy

Paolo Zamboni, MD is a member of the following medical societies: American Venous Forum and New York Academy of Sciences

Disclosure: Nothing to disclose.

Chief Editor

John Geibel, MD, DSc, MA  Vice Chair and Professor, Department of Surgery, Section of Gastrointestinal Medicine, and Department of Cellular and Molecular Physiology, Yale University School of Medicine; Director, Surgical Research, Department of Surgery, Yale-New Haven Hospital

John Geibel, MD, DSc, MA is a member of the following medical societies: American Gastroenterological Association, American Physiological Society, American Society of Nephrology, Association for Academic Surgery, International Society of Nephrology, New York Academy of Sciences, and Society for Surgery of the Alimentary Tract

Disclosure: AMGEN Royalty Consulting; ARdelyx Ownership interest Board membership

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A 90-year-old man referred with abdominal wall abscess in the right upper quadrant.
Computed tomography (CT) scan demonstrating a grossly inflamed gallbladder with a stone within the gallbladder, with partial gallbladder herniation into overlying abdominal wall musculature, marked overlying tissue inflammation, and fistulous tract.
Table. Case Reports of Cholecystocutaneous Fistula in the Past 50 Years From 1961 to 2011
Author(s)Year PublishedNumber of CasesCountry of Origin
Gordon et al[6] 20111United States of America
Sayed et al[7] 20101United Kingdom
Pezzilli et al[8] 20101Italy
Metsemakers et al[9] 20101Belgium
Tallon Aquilar et al[10] 20101Spain
Hawari et al[11] 20101United Kingdom
Gandhi et al[12] 20091New Zealand
Murphy et al[13] 20081United Kingdom
Ijaz et al[14] 20081United Kingdom
Chatterjee et al[15] 20071India
Malik et al[16] 20071United Kingdom
Nagral et al[17] 20071India
Marwah et al[18] 20071India
Shrestha et al[19] 20061United Kingdom
Cruz et al[20] 20061Brazil
Salvador-Izquierdo et al[21] 20061Spain
Yuceyar et al[22] 20051Turkey
Khan et al[23] 20051Saudi Arabia
Dutriaux et al[24] 20051France
Gossage et al[25] 20041United Kingdom
Vasanth et al[26] 20041United States of America
Mathonnet et al[27] 20021France
Chang et al[28] 20021Taiwan
Flora et al[29] 20011United Kingdom
Ramos Rincon et al[30] 20011Spain
Nicholson et al[31] 19991United States of America
Avital et al[32] 19981Israel
Kumar[33] 19981United States of America
Andley et al[34] 19961India
Birch et al[35] 19911United Kingdom
Carragher et al[36] 19901United Kingdom
Rosario et al[37] 19901United States of America
Sevonius et al[38] 19881Sweden
Gibson et al[39] 19871United Kingdom
Bilanovic et al[40] 19871Croatia
Tuna et al[41] 19861United States of America
Hakaim et al[42] 19861United States of America
Rye et al[43] 19851Denmark
Kulicki et al[44] 19841Poland
Davies et al[45] 19841United Kingdom
Abril et al[46] 19841United States of America
Nayman[47] 19831Australia
Ulreich et al[48] 19831United States of America
Hoffman et al[49] 19821United States of America
Fitchett et al[50] 19701United States of America
Callen[51] 19791United States of America
Orr[52] 19791Australia
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