Type II Gall Bladder Perforation with Abdominal Wall Abscess in a Cirrhotic Patient: Case Report and Review of the Literature

CASE REPORTS

Type II Gall Bladder Perforation with Abdominal Wall Abscess in a Cirrhotic Patient: Case Report and Review of the Literature Murad Aljiffry, MD,* Mark Walsh, MD,* Kevork Peltekian, MD,† and Michele Molinari, MD* *Department of Surgery and †Department of Medicine, QEII Medical Center, Halifax, Nova Scotia, Canada Gallbladder perforation is a rare condition even in patients affected by acute cholecystitis. Most patients who present with gallbladder perforations are diagnosed preoperatively by radiologic imaging. Morbidity and mortality for this condition has improved significantly over the last few decades. We encountered a cirrhotic patient who was affected by primary sclerosing cholangitis and who developed a type II gallbladder perforation before undergoing a cadaveric liver transplantation. In this article, we report her clinical course and our review of the current literature on this rare condition. (J Surg 65:367-371. Crown Copyright © 2008 Published by Elsevier Inc. on behalf of the Association of Program Directors in Surgery.) KEY WORDS: cholecystitis, gallbladder perforation, liver fail-

ure, liver transplantation, cholelithiasis COMPETENCY: Patient Care

INTRODUCTION In the adult population, 10% to 20% of the individuals are affected by asymptomatic cholelithiasis.1 Among them, 1% to 2% will suffer from acute cholecystitis in their lifetime.2 Perforation of the gallbladder is a rare complication that occurs in 2% to 11% of all cases of acute cholecystitis,3,4 and it represents a surgical challenge because of the delay in recognition and treatment.5,6 Gallbladder perforations are classified into 3 categories7: type I includes patients with free perforation in the peritoneal cavity, type II includes those with localized perforation, and type III consists of cholecysto-enteric fistulas (Fig. 1). We present a case of a woman with primary sclerosing cholangitis who presented with a large phlegmon of her abdominal wall caused by type II gallbladder perforation while waiting for a cadaveric liver transplant. Correspondence: Inquiries to Michele Molinari, MD, Department of Surgery, Dalhousie University, 1278 Tower Road, Room 6-254, Solid Organ Transplantation, Halifax, Nova Scotia, Canada B3H 2Y9; fax: 001-902-473-7639; e-mail: michelemolinari67@hotmail. com

CASE REPORT A 59-year-old woman with primary sclerosing cholangitis was referred for liver transplantation as she developed ascites, jaundice, and spontaneous bacterial peritonitis. An initial abdominal computerized tomography (CT) showed the presence of a contracted gallbladder with thickened wall that contained 2 calcified stones. Her medical and surgical history was remarkable for type II diabetes, arterial hypertension, and transabdominal hysterectomy for fibromas. Her medications included ursodeoxycholic acid, nadolol, furosemide, spironolactone, and lactulose. She was listed for cadaveric liver transplantation for Child-Pugh C decompensated cirrhosis (MELD score 22), and a few months later, she presented to her local emergency room with severe abdominal pain, fever (38.9°C), and a 10-cm phlegmon in the right side of her abdominal wall (Fig. 2). A repeat CT scan revealed the presence of severe pericholecystic inflammatory reaction, which consisted of a perforated gallbladder wall at the fundus with a 1.8-cm gallstone wedged in the rectus abdominus muscle surrounded by a phlegmon of the anterior abdominal wall (Fig. 3). On admission to our medical center, she appeared in distress, febrile (37.8°C), hypotensive (92/52 mm Hg), and tachycardic (102 HR/min). Her medical condition improved after parenteral broad spectrum antibiotics. Percutaneous cholecystostomy drainage was attempted without success. Because of her persistent symptoms, she was taken to the operating room for an open cholecystostomy and stone extraction. The extensive inflammatory reaction caused by the gallbladder perforation with over-imposed Enterococcus faecium infection prevented the creation of a surgical cholecystostomy. Therefore, a partial cholecystectomy was performed with extraction of the extraluminal gallstone. She was discharged 3 weeks later, and readmitted a few weeks after for drainage of a subhepatic abscess. Three months later, she was identified as the recipient of a cadaveric liver graft. Her operation was complicated by the presence of extensive inflammation and adhesions between her abdominal wall and the ascending colon. Because

Journal of Surgical Education • Crown Copyright © 2008 Published by Elsevier Inc. on behalf of the Association of Program Directors in Surgery

1931-7204/08/$30.00 doi:10.1016/j.jsurg.2008.07.004

367

FIGURE 1. Graphical representation of all three types of gallbladder perforations.

of the degree of portal hypertension and the severe inflammatory reaction from her previous cholecystectomy, she required massive blood transfusions during her liver transplant that caused hypothermia and secondary coagulopathy. After the vascular anastomoses were completed, the patient was transferred to the intensive care unit to correct her metabolic disarrangement, leaving a temporary silastic biliary drainage in the common bile duct of the graft. During the following 12 hours, her core body temperature and coagulopathy persisted, and she was taken 368

back to the operating room for warm intraperitoneal lavage without any clinical improvements. She expired a few hours after returning to the intensive care unit from multiple organ failure.

DISCUSSION The primary origin of acute cholecystitis is the persistent occlusion of the cystic duct by the presence of an impacted stone that causes increased gallbladder wall tension, epithelial injury, re-

Journal of Surgical Education • Volume 65/Number 5 • September/October 2008

FIGURE 2. Intraoperative picture of the patient’s abdominal wall before undergoing partial cholecystectomy and removal of an impacted gallstone from the abdominal rectus muscle. Of particular interest is the noticeable right subcostal phlegmon that was excised during surgery (Black arrow).

lease of phospholipases, degradation of adjacent cell membranes, and intense inflammatory reaction.8 In only 2% to 30% of patients, the disease progresses to gangrenous cholecystitis with wall perforation.9 For most patients, the impacted stone dislodges spontaneously, and the clinical conditions improve with supportive care.10 We described a rare presentation of type II gallbladder perforation in a woman with Child-Pugh C cirrhosis. To our knowledge, no similar cases have ever been reported beforehand. It is well known that physicians who care for cirrhotic individuals have to take in consideration patients’ liver function before proposing any therapeutic options. In 1964, Child and Turcotte11 validated a clinical instrument to stratify the perioperative risks of cirrhotic individuals who undergo surgical shunts for portal vein decompression in 3 categories: A (low risk), B (medium risk), and C (high risk). In 1973, this classification was improved by Pugh et al12 who extended the variables by including serum level of bilirubin, albumin, international normalized ratio (INR), presence or absence of ascites, and encephalopathy (Table 1). Each variable contributes equally to determine the degree of hepatic dysfunction. The postoperative mortality is predicted by the Child-Pugh’s classification in a linear correlation. For major surgical procedures, class A patients have perioperative mortality that ranges between 5% and 15%, class B between 16% and 30%, and class C between 90% and 95%.13 During the last few decades, morbidity and mortality of noncirrhotic patients affected by gallbladder perforation have improved because of better diagnostic modalities, preoperative care, postoperative care, and surgical techniques.14,15 However, our systematic review of the English medical literature (from 1950 until May 2008) has shown that no studies exhibit the best management of cirrhotic patients affected by gallbladder perforation, as the only published report on clinical outcomes of 31 individuals affected by this condition did not include any cirrhotic patients16 (Table 2). It is well known that the man-

agement of cholelithiasis in the presence of Child-Pugh C cirrhosis is a complex issue, and interventions to decrease the perioperative risks of these patients have not shown to be effective in randomized trials.19 Edema of the gallbladder wall, leucopenia caused by hypersplenism, and the presence of ascites that predispose to spontaneous bacterial peritonitis make the diagnosis of gangrenous cholecystitis more difficult than in the general population.17 Gallstones occur twice as frequently in cirrhotic patients as they do in noncirrhotic individuals1; among cirrhotic patients, those with Child-Pugh C have the highest prevalence.20 This finding has been attributed to several factors, such as hemolysis caused by hypersplenism, reduction in biliary acidity, functional alterations in the gallbladder, and metabolic liver failure, which results in an increase in unconjugated bilirubin secretion.21 Individuals with Child-Pugh class A and B cirrhosis who suffer from cholecystitis are safely managed by cholecystectomy with minimal morbidity and mortality.21,22 During the last decades, laparoscopic cholecystectomy has become the treatment of choice for these patients because it is associated with less blood loss, shorter operative time and hospitalization, and decreased intra-abdominal adhesions compared with open cholecystectomy.22 Much more controversial is the management of Child C individuals as data are lacking to support decisions on when to intervene and what therapies to adopt. Child-Pugh C cirrhotic patients have an expected survival rate of only 45% at 1 year without liver transplantation. As liver trans-

FIGURE 3. Computerized tomography (CT) of the abdomen and pelvis with oral and intravenous contrast. There is evidence of gallbladder perforation with extensive phlegmonous changes (Arrow A) in the perihepatic space and overlying the abdominal wall. Two calcified stones are appreciated in the gallbladder fossa. One is impacted in the cystic duct (Arrow B) and the second stone is lodged outside the gallbladder between the fundus and the abdominal wall (Arrow C).

Journal of Surgical Education • Volume 65/Number 5 • September/October 2008

369

TABLE 1. Child-Pugh Classification Variable

1 Point

2 Points

3 Points

Total serum bilirubin (mg/dl) Serum albumin (g/l) INR Ascites Encephalopathy

⬍2 ⬎35 ⬍1.7 Absent Absent

2–3 28–35 1.71–2.2 Controlled with medications Grade I–II (or controlled with medications)

⬎3 ⬍28 ⬎2.2 Refractory Grade III–IV (or refractory)

Child-Pugh class A: 5– 6 points. Child-Pugh class B: 7–9 points. Child-Pugh class C: 10 –15 points.

plantation has become the only therapeutic option to improve their survival, it is important to avoid abdominal operations if possible to prevent adhesions that make liver transplantation more challenging. Therefore, for asymptomatic gallstones in cirrhotic patients, consensus confirms that prophylactic cholecystectomy is not indicated.1,23 When compared with uncomplicated acute cholecystitis, gangrenous cholecystitis has increased morbidity and mortality.24 Therefore, emergent cholecystectomy is advocated.25 In a recent meta-analysis of Child-Pugh A and B cirrhotic patients who underwent laparoscopic cholecystectomy, the overall morbidity rate was found to be 21% compared with 8% (p ⫽ 0.05) for the general population with the overall mortality not statistically different for the 2 groups (0.59% vs 0.13%).22 However, several reports have shown that in the presence of Child-Pugh C cirrhosis, the mortality rate after cholecystectomy for acute cholecystitis is as high as 17% to 25%.22,26 For this reason, less invasive treatments, such as percutaneous gall bladder aspiration and cholecystostomy drainage, have been recommended.27 Although percutaneous gall bladder aspiration seems a very promising technique to provide quick resolution of symptoms and can be safely repeated over time when necessary, long-term results are not currently available.27 The advantages of gallbladder aspiration in Child-Pugh C cirrhotic patients who present with acute cholecystitis are several, as this technique does not require general anesthesia and can be performed at the bedside. However, the presence of ascites, coagulopathy, and encephalopathy that are present in decompensated cirrhotic patients are contraindications for this technique.

Another minimally invasive approach is decompression of the inflamed gall bladder by percutaneous cholecystostomy drainage.28 This effective modality has been recommended for individuals who are not fit for surgery. The benefits of cholecystostomy drainage have to be weighed against the risks of recurrent cholecystitis that occurs in 25% to 30% after removal of the cholecystostomy tube.29,30 In addition, patients with severe ascites may not be candidates for this procedure as they often develop ascitic fluid leak and have a significant increased risk of secondary peritoneal bacterial infection from contamination of percutaneous drainages.

SUMMARY We presented a case of a 59-year-old woman with Child-Pugh C cirrhosis who survived partial cholecystectomy for type II gallbladder perforation and who died 2 months later from complications that occurred during cadaveric liver transplantation. Currently, no data are available on morbidity and mortality of perforated gallbladder in Child-Pugh C cirrhotic patients and on the long-term consequences of this condition for individuals who are candidates to liver transplantation. This article is the only report currently available in the medical literature. The final outcome of our patient would suggest that liver transplantation after gallbladder perforation is a very high risk procedure; the inflammatory response surrounding the hepatic hilum makes the operation extremely challenging. As adhesions and inflammatory response decrease over time, it may be prudent to delay listing patients for liver transplantation for several months

TABLE 2. Summary of Reported Outcomes for Cholelithiasis Extracted by Systematic Review of the Current Literature from EMBASE and MEDLINE Digital Medical Databases (1950 until May 2008) Reference(s)

Variable(s)

Value(s)

1 2 3, 4, 14 17 17

Adult population affected by asymptomatic cholelithiasis Percentage of patients affected by cholelithiasis developing cholecystitis Percentage of patients affected by cholecystitis developing gallbladder perforation Morbidity associated with elective laparoscopic cholecystectomy in the general population Morbidity associated with elective laparoscopic cholecystectomy in Child-Pugh class A and B cirrhotic patients Mortality associated with elective laparoscopic cholecystectomy in the general population Mortality associated with elective laparoscopic cholecystectomy in Child-Pugh class A and B cirrhotic patients Mortality associated with urgent cholecystectomy in Child-Pugh class A and B cirrhotic patients for cholecystitis

10% to 20% 1% to 2% 2% to 30% 8% 21%

17 17 17, 18

370

0.13% 0.59% 17% to 25%

Journal of Surgical Education • Volume 65/Number 5 • September/October 2008

after the development of perforated gangrenous cholecystitis if possible, even in the presence of a complete clinical recovery. Although this recommendation is based only on 1 observation, our experience would suggest that this approach might allow the severe inflammatory response to resolve and make the liver transplant surgery safer. As gallbladder perforation is a rare condition, trials on the best management of cirrhotic patients affected by this disease would be very difficult to carry out even if extremely needed.

REFERENCES 1. Gibney EJ. Asymptomatic gallstones. Br J Surg. 1990;77:

368-372. 2. Friedman GD. Natural history of asymptomatic and

symptomatic gallstones. Am J Surg. 1993;165:399-404. 3. Abu Dalu J, Urca I. Acute cholecystitis with perforation

sonography and percutaneous treatment. Br J Radiol. 1993;66:1052-1054. 16. Menakuru SR, Kaman L, Behera A, et al. Current man-

agement of gall bladder perforations. ANZ J Surg. 2004; 74:843-846. 17. Silva MA, Wong T. Gallstones in chronic liver disease.

J Gastrointest Surg. 2005;9:739-746. 18. Cucinotta E, Lazzara S, Melita G. Laparoscopic cholecystec-

tomy in cirrhotic patients. Surg Endosc. 2003;17:1958-1960. 19. Xu Q, Gu L, Wu ZY. Operative treatment for patients

with cholelithiasis and liver cirrhosis. Hepatobiliary Pancreat Dis Int. 2007;6:479-482. 20. Fornari F, Imberti D, Squillante MM, et al. Incidence of

gallstones in a population of patients with cirrhosis. J Hepatol. 1994;20:797-801.

into peritoneal cavity. Arch Surg. 1971;102:108-110.

21. Bloch RS, Allaben RD, Walt AJ. Cholecystectomy in pa-

4. MacDonald JA. Perforation of the gallbladder associated

tients with cirrhosis. Arch Surg. 1985;120:669-672.

with acute cholecystitis. Ann Surg. 1966;164:849-852.

22. Puggioni A, Wong LL. A meta-analysis of laparoscopic

5. Fletcher AG, Ravdin IS. Perforation of the gall bladder.

Am J Surg. 1951;81:178-185. 6. Lennon F, Green WE. Perforation of the gall bladder. J R

Coll Surg Edinb. 1983;28:169-173. 7. Niemeirer DW. Acute free perforation of the gall bladder.

Am Surg. 1934;99:922-944. 8. Fagan SP, Awad SS, Rahwan K, et al. Prognostic factors

for the development of gangreneous cholecystitis. Am J Surg. 2003;186:481-485. 9. Bedirli A, Sakrak O, Soziier EM, et al. Factors effecting

cholecystectomy in patients with cirrhosis. J Am Coll Surg. 2003;197:921-926. 23. Orozoco H, Takahashi T, Mercado MA, et al. Long-term

evaluation of asymptomatic cholelithiasis diagnosed during abdominal operation for variceal bleeding in patients with cirrhosis. Am J Surg. 1994;168:232-234. 24. Morfin E, Ponka JL, Brush BE. Gangrenous cholecystitis.

Arch Surg. 1968;96:567-572. 25. Wison AK, Kozol RA, Salwen WA, et al. Gangrenous cho-

lecystitis in an urban VA hospital. J Surg Res. 1994;56:402404.

the complications in the natural history of acute cholecystitis. Hepatogastroenterology. 2001;48:1257-1278.

26. Aranha GV, Sontag SJ, Greenlae HB. Cholecystectomy in

10. Croley GG. Gangrenous cholecystitis. Am Surg. 1992;58:

cirrhotic patients: a formidable operation. Am J Surg. 1982;143:55-60.

284-292. 11. Child CG, Turcotte JG. Surgery and portal hypertension.

Philadelphia, Pa: Saunders, 1964:50-64. 12. Pugh RNH, Murray-Lyon IM, Dawson JL, et al. Tran-

section of the esophagus in the bleeding esophageal varice. Br J Surg. 1973;60:648-652. 13. Tsai MH, Chen YC, Ho YP, et al. Organ System Failure

scoring system can predict hospital mortality in critically ill cirrhotic patients. J Clin Gastroenterol. 2003;37:251-257. 14. Ong CL, Wong TH, Rauff A. Acute gall bladder

perforation – a dilemma in early diagnosis. Gut. 1991; 32:956-958. 15. Longo JM, Bilba JI, Devilla VH, et al. Gall bladder per-

foration and bile leakage: diagnosis by color Doppler

27. Currò G, Cucinotta E. Percutaneous gall bladder aspira-

tion as an alternative to laparoscopic cholecystectomy in Child-Pugh C cirrhotic patients with acute cholecystitis. Gut. 2005;08:898. 28. Patel M, Miedema BW, James MA, Marshall JB. Percutane-

ous cholecystostomy is an effective treatment for high-risk patients with acute cholecystitis. Am Surg. 2000;66:33-37. 29. Sugiyama M, Tokuhara M, Atomi Y. Is percutaneous cho-

lecystostomy the optimal treatment for acute cholecystitis in the very elderly? World J Surg. 1998;22:459-463. 30. Van Steenbergen W, Ponette E, Marchal G, et al. Percu-

taneous transhepatic cholecystostomy for acute complicated cholecystitis in elderly patients. Am J Gastroenterol. 1990;85:1363-1369.

Journal of Surgical Education • Volume 65/Number 5 • September/October 2008

371