Cholecystocutaneous Fistula


Article Author:
Abdul Waheed
George Mathew


Article Editor:
Faiz Tuma


Editors In Chief:
Kranthi Sitammagari
Mayank Singhal


Managing Editors:
Avais Raja
Orawan Chaigasame
Carrie Smith
Abdul Waheed
Khalid Alsayouri
Trevor Nezwek
Radia Jamil
Erin Hughes
Patrick Le
Anoosh Zafar Gondal
Saad Nazir
William Gossman
Hassam Zulfiqar
Hussain Sajjad
Steve Bhimji
Muhammad Hashmi
John Shell
Matthew Varacallo
Heba Mahdy
Ahmad Malik
Sarosh Vaqar
Mark Pellegrini
James Hughes
Beata Beatty
Beenish Sohail
Nazia Sadiq
Hajira Basit
Phillip Hynes


Updated:
8/4/2019 9:58:34 AM

Introduction

A fistula is known in the medical field as an abnormal communication between two epithelial surfaces. This abnormal communication can result as a complication of a disease or surgical intervention.  The naming of the fistula follows the two surfaces or lumens it connects[1]. A cholecysto-cutaneous fistula is, therefore, abnormal communication between the gallbladder and the skin. Similarly, entero-cutaneous fistula is an abnormal communication between the small bowel and the skin.

Cholecysto-cutaneous fistula is a rare clinical entity in the surgical practice, nowadays. Reports of spontaneous cholecysto-cutaneous fistulae occurring as early as 17th century were registered. The condition develops as a result of a complication from neglected calculous cholecystitis. However, it occurs extremely infrequently in the current surgical practice. With the advancement in diagnostic imaging, biliary tract diseases are diagnosed early nowadays. The availability of efficient and safe surgical treatment made the complication like fistula extremely uncommon.[2][3][4] 

Etiology

Cholecysto-cutaneous fistulas present as an uncommon complication of neglected calculous cholecystitis. This condition has also been associated with acalculous cholecystitis or carcinoma of the gall bladder. A significant proportion of patients with this condition present with nonspecific symptoms and rarely disclose a history of previous gallbladder disease.[5] Once the gallbladder wall is inflamed, it starts to necrosis if left untreated. Complete wall necrosis results in leaking of the bile into the adjacent tissue. One of the potential sites of leaking is the abdominal wall and eventually the covering skin resulting in cholecysto-cutaneous fistula. 

Epidemiology

Due to the rarity of the condition, the available information are limited. The affected patients are usually older females. When questioned, patients may mention previous episodes of right upper quadrant pain. This pain hints at gallstone disease. The most common site of the fistulous opening is the right upper quadrant of the abdomen. Painless, draining fistulas associated with this condition have been reported in many locations on the abdominal wall such as right upper quadrant, right iliac fossa, umbilicus, right lumbar region, left lower quadrant, and even as far as the gluteal region. Gallbladder fistulas have been known to occur following percutaneous treatment for acute cholecystitis and reoccurrence of cholecystitis with the fistula forming along the previous track of the cholecystostomy drain.[6][7]

Pathophysiology

Obstruction of the biliary outflow by a gallstone is thought to play a significant role in the pathophysiology of the development of this condition. Obstruction of the cystic duct leads to increased intra-gallbladder pressure. Unrelieved obstruction to the bile outflow leads to compromise of gallbladder wall blood circulation as well as lymphatic drainage which eventually results in necrosis of the gallbladder wall. An open perforation of the gallbladder pericholecystic abscess follows. Once perforated, the gallbladder may drain into the peritoneal cavity, localized causing Perito-ni localized abscess. This abscess can then develop into an external fistula due to its adherence to the abdominal wall. It expels its contents through an area of least resistance or shortest route. Conditions such as polyarteritis nodosa, typhoid, trauma, or drug treatments such as steroids, may be predisposing factors. Carcinoma of the gallbladder is known to present with an external fistula. Cutaneous gallbladder fistula is usually a late sequelae of chronic biliary tract disease. It has also been reported after inadequate treatment with acute cholecystitis

History and Physical

The clinical presentation may be variable depending on the associated pathology, the age of the patient, and the presence or absence of an associated abdominal wall abscess. The typical clinical presentation is that of a patient's sinus persistently discharging bile. There is usually a history of chronic biliary tract disease even though patients may not report it as a primary complaint. At times the patient may give a history of the expulsion of gallstones through the fistula. The site of the external opening of the fistula is variable and depends on the anatomical course. The commonest site of the outer opening is the right upper quadrant of the abdominal wall. However, the opening of the fistula tract may be present in the left costal margin, right iliac fossa, right groin, or right gluteal region. Patients may be toxic when there is an acute infection or associated abscess. The character of the discharge from the fistula may vary according to the underlying pathology. A lump may be palpable deep to the fistula due to the underlying inflamed or malignant gallbladder.

Evaluation

Ultrasonography is the first modality of imaging in the background of gallstone disease, but it has not been found to be of significant use in gallbladder fistula even though it demonstrates the presence of gallstones reliably. CT scan is the most useful imaging modalities for diagnosing this complication. CT imaging will reveal evidence of gallstones and communication to the gallbladder. A CT fistulogram can demonstrate the fistula connecting to the gallbladder and biliary tract which is contributory to the establishment of this diagnosis. A CT fistulogram can be achieved by injecting diluted contrast through the fistulous tract while performing the CT. A CT fistulogram helps to delineate the course of the track accurately and establish the anatomical relationship of this tract. This imaging modality also helps excludes any internal fistula, and other associated pathology. A clear delineation of the fistulous tract helps in planning the approach and extent of surgery. Routine blood tests may not be helpful unless there is an abscess collection. MRI with Cholangiopancreatography (MRCP) has also been found to be useful in establishing the diagnosis. [8][9]

Treatment / Management

The management of cholecyst-cutaneous fistula requires initial drainage of any associated abscess and administration of appropriate antibiotics. If there is an associated mass, it is expedient to obtain a cytologic diagnosis by fine-needle aspiration cytology (FNAC) to exclude an underlying malignancy. If the cytology is negative, or there is no suspicion of malignancy, the next step to follow is an elective cholecystectomy once the patient is stable and his or her general condition optimized. An open cholecystectomy with excision of the fistulous tract is the definitive treatment. Laparoscopic cholecystectomy can be considered to reduce the morbidity since many of these patients will be older with multiple associated comorbidities. Percutaneous treatment with removal of the gallstones only is reserved for the elderly with multiple comorbidities, or for those who are too debilitated to withstand surgery. Before the definitive surgery is performed, an effort must be taken to ensure that there is no associated bile duct obstruction or stones. An indication of an unresectable disease is if a gallbladder fistula occurs in the setting of cancer with an associate dated mass. In this case, the overall outcome is dismal because gallbladder cancer has a predilection for seeding and spreading to the adjoining anatomical structures.[10][11]

Complications

  • Necrotizing fasciitis
  • Skin irritation
  • Malignant changes

Pearls and Other Issues

In any elderly patient who has a fistula discharging bile on the right abdominal wall, practitioners should consider the possibility of a cholecysto-cutaneous fistula. It is possible a fistula formed in a patient who underwent previous percutaneous treatment of an acute cholecystitis and has recurrent cholecystitis. Such patients should be investigated adequately for underlying pathology and associated biliary tract disease.

Enhancing Healthcare Team Outcomes

Cholecysto-cutaneous fistulas are rare today but like any cutaneous fistula need proper drainage and skin protection, while at the same time managing the primary cause. Specialized would care nurses usually provide day to day management of cholecysto-cutaneous fistulas in the same manner as any cutaneous fistula by recording the amount of drainage, characteristics of the drainage and at the same time providing skin protection. The results should be reported to the surgeon. If abnormal, such as slow drainage, pus, or signs of infection, the surgeon should be contacted immediately. The nurse should monitor the patient and provide family support including ongoing education of the patient and family. The prognosis for benign causes is good as long as the primary condition is managed. However, in patients with a malignant cause, the prognosis is guarded and the fistula may not be treatable. It is important for healthcare workers to know that any chronic wound that is left untreated can develop malignant changes after 10-20 years, hence close monitoring by the interprofessional team is necessary The best outcomes are achieved by interprofessional teamwork in treating these challenging cases. [9][8][12]


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Cholecystocutaneous Fistula - Questions

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A 72-year-old male presents to the emergency department with a 3 cm tender and erythematous lump at the right upper quadrant present for 2 weeks. The patient is admitted and surgery is planned for the morning but during the night there is spontaneous drainage of a gallstone and purulent drainage. He is diagnosed with a cholecystocutaneous fistula. Fistulogram confirms the diagnosis. The family is hesitant to consent to surgery as the patient has COPD, CAD, and CHF. Select the best option for treatment.



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Which of the following factors is not contributory for the development of a cholecyst cutaneous fistula?



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An 80-year-old woman presents with a sinus on the right upper quadrant of the abdominal wall that is discharging bile. She gives a history of extruding a few small gallstones through the sinus. Your clinical suspicion is a gallbladder fistula. Which investigation would best confirm the diagnosis and help plan the treatment?



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A 78-year-old male presents to the hospital with a persistent right upper quadrant pain for several days. He developed a small draining area of clear green discharge at Murphy's spot on the right upper quadrant. After a clinical and ultrasound assessment, a cholecysto-cutaneous fistula is diagnosed. He has a low-grade fever, normal blood pressure, and normal temperature. Which of the following is the next best step in the management of this patient?



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For the past 8 days, a 65-year-old female is being treated for urosepsis in addition to her uncompensated heart failure and uncontrolled diabetes. Today, she developed right upper quadrant pain and tenderness. Ultrasound examination of the biliary system identified a thickened wall with edema and possible pericholecystic fluid. Which of the following is the most suitable treatment of this condition?

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An 83-year-old male developed a cholecysto-cutaneous fistula after prolonged untreated cholecystitis. He has mild pain and asymptomatic otherwise, except the drainage site at the right upper quadrant. Which of the following is the next best step in the management of this patient?



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Cholecystocutaneous Fistula - References

References

Schippers HJW,Toorenvliet BR, [A late complication of cholecystitis]. Nederlands tijdschrift voor geneeskunde. 2018 Jun 22     [PubMed]
Wang YF,Chen HQ, Hepatobiliary and Pancreatic: Spontaneous cholecystocutaneous fistula. Journal of gastroenterology and hepatology. 2018 Jun 12     [PubMed]
Rinzivillo NMA,Danna R,Leanza V,Lodato M,Marchese S,Basile F,Zanghì GN, Case Report: Spontaneous cholecystocutaneous fistula, a rare cholethiasis complication. F1000Research. 2017     [PubMed]
Gerrard A,Date R, Delayed Spontaneous Passage of Gallstones via Cholecystocutaneous Fistula. ACG case reports journal. 2017     [PubMed]
Jayasinghe G,Adam J,Abdul-Aal Y, Unusual presentation of gallbladder perforation. International journal of surgery case reports. 2016     [PubMed]
Maynard W,McGlone ER,Deguara J, Unusual aetiology of abdominal wall abscess: cholecystocutaneous fistula presenting 20 years after open subtotal cholecystectomy. BMJ case reports. 2016 Mar 31     [PubMed]
Micu BV,Andercou OA,Micu CM,Militaru V,Jeican II,Bungărdean CI,Mogoantă SŞ,Miclăuş DR,Pop TR, Spontaneous cholecystocutaneous fistula as a primary manifestation of gallbladder adenocarcinoma associated with gallbladder lithiasis - case report. Romanian journal of morphology and embryology = Revue roumaine de morphologie et embryologie. 2017     [PubMed]
Pripotnev S,Petrakos A, Cholecystocutaneous fistula after percutaneous gallbladder drainage. Case reports in gastroenterology. 2014 Jan     [PubMed]
Kapoor Y,Singh G,Khokhar M, Spontaneous cholecystocutaneous fistula-not an old time story. The Indian journal of surgery. 2013 Jun     [PubMed]
Jayant M,Kaushik R,Attri AK, Spontaneous cholecystocutaneous abscess. Indian journal of gastroenterology : official journal of the Indian Society of Gastroenterology. 2014 Sep     [PubMed]
Ozdemir Y,Yucel E,Sucullu I,Filiz I,Gulec B,Akin ML,Yildiz M, Spontaneous cholecystocutaneous fistula as a rare complication of gallstones. Bratislavske lekarske listy. 2012     [PubMed]
Shams C,Cannon M,Bortman J,Hakim S, Stone-Induced Purulent Choledocoduodenal Fistula Presenting with Ascending Cholangitis. ACG case reports journal. 2018     [PubMed]

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