Medical College, Bemina, Srinagar , Jammu and Kashmir, India 2 Department of Surgery, Sheri Kashmir Institute of Medical Sciences

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1 eissn: Original Research Article Shams Ul Bari 1, Ajaz A Malik 2, Shiraz A Rather 2, Asima Khan 3 1 Department of General Surgery, Sheri Kashmir Institute of Medical Sciences Medical College, Bemina, Srinagar , Jammu and Kashmir, India 2 Department of Surgery, Sheri Kashmir Institute of Medical Sciences Soura, Srinagar , Jammu and Kashmir, India 3 Sheri Kashmir Institute of Medical Sciences Medical College,Bemina, Srinagar , Jammu and Kashmir, India Abstract Spontaneous biliary peritonitis may be due to idiopathic rupture of the extra or intra hepatic biliary tract. It is a rare clinical entity and the diagnosis is often delayed due to their nonspecific signs and symptoms. This paper describes the retrospective study conducted at Sher-i-.Kashmir Institute of Medical Sciences Srinagar, over a period of five years (from June 2008 to May 2013) to analyze the risk factors, clues to early diagnosis and tailored interventions in the management of spontaneous biliary peritonitis. Outcome of the present study indicate that with high index of suspicion, in the presence of these risk factors, rapid recognition of peritonitis along with adequate resuscitation and tailored operative interventions, are mandatory to reduce the morbidity and mortality from primary biliary peritonitis. Key Words: Biliary Peritonitis, cholilithiasis, Endoscopic retrograde cholangio pancreatography Cite this article as: Shams Ul Bari, Ajaz A Malik, Shiraz A Rather, Asima Khan. Spontaneous biliary peritonitis- A Retrospective study. American Journal of Advances in Medical Science. 2014; 2(4): Source of Support: Nil, Conflict of Interest: None declared. 18

2 1.0 Introduction Spontaneous biliary peritonitis is a rare cause of acute abdomen pain. In spontaneous biliary peritonitis there is perforation in the wall of the extra-hepatic or intra-hepatic duct occurs without any traumatic or iatrogenic injury and have been described more often in neonates [1]. The symptoms may be acute or insidious delaying the diagnosis. The etiology of spontaneous biliary peritonitis has always remained obscure. Spontaneous biliary peritonitis due to idiopathic rupture of the extra or intra hepatic biliary tract is a rare clinical entity. The diagnosis of biliary tract perforations is often delayed due to their nonspecific signs and symptoms and no antecedent history of biliary tract disease in the past. The incidence of gallbladder (GB) perforation in acute cholecystitis is 3-10% in adults and the risk factors include age more than 60 years, immunosuppression, steroid use, and severe systemic disease [1a]. Perforation of common bile duct (CBD) is even a rarer entity in adults with only very few cases having been reported earlier, with cholelithiasis, choledocholithiasis and tumor obstruction of ampulla as possible etiologies of perforation [2]. 2.0 Material and methods The present study was a retrospective study involving seventeen patients with an average of 3 patients per year (range 2-4) admitted to accident and emergency department of Sher-i-.Kashmir Institute of Medical Sciences Srinagar, over a period of five years (from June 2008 to May 2013). Patient demographic profile, presentation, course of events, laboratory findings and operative techniques were evaluated retrospectively by studying the case sheets of the patients stored in the department of medical records of hospital. Patients were put on conservative line of management to which they did not respond. Then the patients were suggested for complete blood count, serum amylase, kidney and liver function tests, X-ray chest/abdomen, ultrasonography (USG) in all and computed tomography (CT) scan in selected patients where the USG did not reveal sufficient information. The USG revealed calculus in common bile duct in eleven patients and all the patients were taken for preoperative Endoscopic Retrograde Cholangio Pancreatography (ERCP). ERCP failed to provide any relief in two of the patients. Despite ERCP, the condition worsened over a period of time in all the patients and evolved in either frank or localized peritonitis (suggested by clinical, biochemical and radiological evidence) of unknown etiology. Laproscopy was not possible due to non-availability of laparoscope in emergency in the study area. All the patients were operated with a diagnostic dilemma via a midline or right para-median incision which was extended upwards, after excluding the perforated appendix or any other gut perforation. Diagnosis of spontaneous biliary perforation was made during laparotomy and the procedures were tailored individually. Cholecystectomy with CBD exploration and repair over a T- tube was performed in 2 patients where ERCP had failed. However cholecystectomy alone was done in 19

3 remaining patients 15 patients. After a thorough peritoneal lavage, the right sub hepatic area was drained and adequate antibiotic coverage was instituted. T-tube cholangiogram was done on 8-10 th postoperative day and the T-tube was removed in case of no residual calculus in CBD. As the present study was a retrospective study, follow up of the efficacy of treatment was not possible. Statistical analysis was carried out in accordance with the small size of sample by an expert statistician using SPSS 17 statistics package (Chicago, IL). Normal Deviate test (Z- test) was used, P value less than 0.05 was considered as statistically significant level. 3.0 Results and discussion On evaluating the case sheets of the patients, it was found that the patients were admitted with complaint of pain of variable severity in upper abdomen. Results are summarized in Table-1. The mean age of patients was years with a standard deviation of ±18.5 (median-72.0, range 16 to 93 years), and male- female ratio of 1:7.5 (2 males to 15 females). Table-1: Clinico-epidemiological profile of the patients Parameter Present/absent (%) P-value Female sex 15/2 (88.23) Obesity 15/2 (88.23) Bilious PL 6/11 (35.3) Increased ALP 14/3 (82.4) Billirubin >2 12/5 (70.6) Increased TLC 10/7 (58.8) Dilated CBD 17/0 (100) Choledocolithiasis 11/6 (64.7) Morbidity 8/9 (47) %-percentage, PL-peritoneal lavage, ALPalkaline phosphatase, TLC-total leukocyte count, CBD-common bile duct. Fifteen out of 17 patients were obese with BMI more than 30 (88.23%, P=0.000, median BMI 33). Because of vague presentation and non-specific clinical features, an average delay of diagnosis was 4 days (range 2 to 6 days).all patients presented with pain of varied severity in the upper abdomen which ultimately culminated in localized (58.82%), or diffuse (41.17%) peritonitis with systemic features like tachycardia, tachypnea, fever or septic shock. None of the patients had the antecedent history of acute or chronic cholecystitis (biliary colic) in the past. Abdominal radiograph revealed no gas under diaphragm while as some USG and CT scan findings seemed to be helpful like presence of free fluid or localized collection in right hypochondrium, partially collapsed gallbladder, and dilated common bile duct (more than 1cm) along with choledocholithiasis. Peritoneal tap was not always diagnostic because bilious aspirate was rarely obtained and instead purulent aspirate was obtained in 11 (64.7%, p=0.086) patients. Raised ALP was seen in 14 (82.35%, P=0.000), bilirubin >2mg/dl in 12 (70.58%, p=0.016), and leukocytosis was seen in 10 (58.82%, p=0.226) patients. Serum amylase was normal in all patients. The perforations were seen in gallbladder in 14 (82.35%) patients, which were single or multiple in number and often seen sealed at the time of exploration. Dilated CBD >1cm was seen in all the 17 patients (P=0.000), while choledocholithiasis was only seen in 11 (64.7%, P=0.086) patients. However no gross perforation in GB or CBD could be observed in 3 20

4 (17.64%) patients. The major post operative complication were septicemia with cholangitis (35.29%), acute respiratory distress syndrome (23.52%), wound infection (17.64%), pulmonary thromboembolism and biliary leak (11.76% each) which emerged as major cause of morbidity. Naked eye examination of the gall bladders showed patchy thinning of the wall of gallbladder. The most common histopathological finding in our study was chronic cholecystitis, 92.3% specimens reported chronic inflammation with mucosal ulceration, denudation, and metaplasia to dysplasia and wall infiltration by chronic inflammatory cells like neutrophils, macrophages, plasma cells and varying degrees of fibrosis. In severe cases mucosal and mural necrosis was seen along with infiltration by neutrophils. Biliary tract perforation most commonly occurs after surgery, trauma or instrumentation. Peritonitis requiring surgical intervention is caused by perforated peptic ulcer in 46% cases (duodenum: gastric: 3:1), appendicitis in 20%, gangrene of the small bowl or gall bladder in 15%, post operative complication in 10%, and miscellaneous causes in 15% cases [3]. Primary biliary peritonitis is a rare clinical entity and has no evident precipitants. Perforation of the biliary tract secondary to rupture of GB (empyema or gangrene) is well documented. The initiating event in majority of these patients is impaction of the stone leading to epithelial injury and ischemia due to GB distention. The site of perforation is at the fundus of GB, which is farthest away from blood supply, or less commonly at the neck of the GB from the pressure of an impacted stone [4]. Spontaneous perforation of CBD is even a rarer entity, more commonly seen in children less than 4 years of age [5]. The proposed etiological factors are choledochal cyst, anomalous union of pancreaticobiliary ductal system, distal bile duct stenosis or atresia leading to congenital weakness of CBD [6,7]. Other rare causes are erosion by tumor, weakness by previous choledochostomy or localized ischemia of CBD wall due to intramural thrombosis, infection or reflux of pancreatic juice [8-10]. Choledocholithiasis with increased intra ductal pressure has also been reported as one of the most important cause of biliary tract perforation[10.] Yet less frequently seen is primary biliary peritonitis without identifiable perforation, and the first such case was reported in 1905 [11]. Explanations for bile escape from imperforate biliary tract include bile transudation across ducts in association with CBD obstruction[12] or solubilisation of normally impermeable bile by trypsin liberated during mild or subacute pancreatitis [13]. Others suggested a possible leak through perforations invisible to naked eye [14]. In the present study, 3 patients with frank bile in peritoneum where no visible perforation were identified and serum amylase was raised in 2 (66%) of them, suggesting the possible role of pancreatitis. However USG did not suggest pancreatitis and CT scan was carried out in all of them. There are reports that in adults, 3-10% cases of acute cholecystitis may have GB perforation and the risk factors for 21

5 adults include immunesuppression, steroid use, and severe systemic disease[1]. Risk factors for primary biliary peritonitis which emerged in retrospective analysis of our cases included age, sex, obesity and the lack of antecedent history of biliary colic or cholecystitis as none of our patients had documented evidence of acute cholecystitis in the past. The infrequency of perforation in the setting of calculus disease of GB is probably due to the thickened wall of the organ that has long been the seat of chronic inflammation. In the present study, 58.82% patients with localized peritonitis and 41.18% with diffuse peritonitis was observed. Due to varied clinical presentation of a rare entity besides lack of sensitive and specific marker, a delay in diagnosis of spontaneous biliary peritonitis is unavoidable. However sonography, scintigraphy and abdominal paracentesis are more commonly used. Sonography shows either generalized ascitis or localized collection of intra peritoneal fluid, and the biliary tree usually is not dilated [13]. Results of scintigraphy and abdominal paracentesis demonstrate that the intraperitoneal fluid originated from biliary tract [14]. In the present study, spontaneous biliary peritonitis was diagnosed using clinical, biochemical and radiological (X-ray abdomen and sonography) findings. Although abdominal paracentesis was performed in all the patients, it was not suggestive of biliary perforation as it mostly revealed frank pus rather than a bilious fluid, due to the superadded infection over a period of time. The interventions performed on patients were influenced by factors like the condition of the patient, site of perforation and the facilities available. However definitive treatment involves surgery which ranges from percutaneous tube drainage [15] to complicated biliary procedures [6]. When there are facilities to confirm diagnosis of perforation of bile duct without operative interventions, percutaneous tube drainage is the ideal treatment and the basis of this is that in most cases the perforation in CBD heals spontaneously as there is usually no distal obstruction[14]. The choice between simple peritoneal drainage and T-tube drainage in perforated CBD is a matter of debate [15]. If a preoperative cholangiogram is possible and shows normal passage of contrast into the duodenum simple peritoneal drainage will suffice, however in case of obstruction or if preoperative cholangiogram is not available, T- tube drainage will be an optimal option as the presumed obstruction causing perforation is due to biliary sludge or insipissated bile, which is expected to clear by itself as the patient recovers from acute condition. In the present work, T tube drainage because of lack of facilities for operative cholangiogram in emergency department. For other conditions encountered as perforations in the gallbladder or in the cystic duct[15], a simple cholecystectomy will cure the patient. In present case, cholecystectomy was done in all patients irrespective of the site of perforation, and CBD exploration with T-tube drainage was performed in patients where ERCP had failed. 22

6 The clinical picture associated with biliary peritonitis varies and the correct preoperative diagnosis is difficult. This, combined with the associated co morbidity of the patient population (usually elderly) significantly contributes to a mortality rate of 30-50% [15]. In the present work it was observed that, in addition to advanced age group and associated co morbidity, a delay in diagnosis (average delay of 4 days) had a significant effect on overall morbidity and mortality (23.53% mortality). 4.0 Conclusion With high index of suspicion, in presence of the risk factors (advanced age, female sex, obesity, asymptomatic cholelithiasis and choledocholithiasis), rapid recognition of peritonitis with adequate resuscitation and tailored operative interventions are mandatory to reduce the morbidity and mortality. Reference 1. Shanahan EF. Spontaneous biliary peritonitis. Br Med J. 1961; 2: a. Petrozza JC, Mastrobattista JM, Monga M. Gallbladder perforation in pregnancy. Am J Perinatol. 1995; 12(5): Piotrowski JJ, Van Stiegmann G, Liechty RD. Spontaneous bile duct rupture in pregnancy. HPB Surg. 1990; 2(3): Crawford E, Ellis H. Generalized peritonitis- The changing spectrum: A report of 100 consecutive cases. Br J Clin Pract. 1985; 39: Kerstein M D, McSwain N E. Spontaneous rupture of the common bile duct. Am J Gastroentrol. 1985; 80: Chardot C, Iskandarani F, De Dreuzy O, Duquesne B, Pariente D, Bernard O. Spontaneous perforation of the biliary tract in infancy: A Series of 11 cases. Eur J Pediatr Surg. 1996; 6(6): Shigland N, Greco R, Rosenfield D. Spontaneous biliary perforation: Does external drainage constitute adequate therapy? J Pediatr Surg. 1996; 33: Rege SA, Lamba S, Sethi H. Spontaneous common bile duct perforation in adult: A Case report and review. Int Surg. 2002; 87: Hill NS, Colapinto ND. Spontaneous perforation of the common bile duct. Br J Surg. 1981; 68: Shanahan EF. Spontaneous biliary peritonitis. Br Med J. 1961; 2: McLaughlin CW. Bile peritonitis. Ann Surg. 1942; 115: Haller JO, Condon VR, Berdon WE, Oh KS, Price AP, Bowen A. Spontaneous perforation of the common bile duct in children. Radiol. 1989; 172: Kasat LS, Borwanker SS, Jain M, Naregal A. Spontaneous perforation of the extrahepatic bile duct in an infant. Pediatr Int. 2001; 17: Spigland N, Greco R, Rosenfeld D. Spontaneous biliary perforation: Does external drainage constitute adequate therapy? J Pediatr Surg. 1996; 31(6):

7 14. Stringel G, Mercer S. Idiopathic perforation of the biliary tract in infancy. J Pediatr Surg. 1983; 18: Borghese M, Caramanico L, Anelli L, De Cesare A, Farrocco G, Spallone G. Etiopathogenetic and physiopathological considerations on biliary peritonitis. Minerva Med J. 1986; 77: