
==== Front
Ann Med Surg (Lond)
Ann Med Surg (Lond)
MS9
Annals of Medicine and Surgery
2049-0801
Lippincott Williams & Wilkins Hagerstown, MD

10.1097/MS9.0000000000000186
00020
3
Case Reports
Spontaneous perforation of the common bile duct in adults presenting as biliary peritonitis: a case report and literature review
http://orcid.org/0000-0001-7831-9845
Bouzid Ahmed MD drbouzidahmed89@gmail.com

http://orcid.org/0000-0001-8885-8818
Fendri Sami MD sami.fendri@hotmail.com

Rejab Haitham MD rejabhaitham@yahoo.fr

http://orcid.org/0000-0003-2291-7250
Trigui Ayman MD ayman.trigui@gmail.com

Mejdoub Youssef MD Youssefmejdoub@outlook.fr

Boujelbene Salah MD salahboujelben5@gmail.com

Department of General Surgery, Habib Bourguiba Hospital, Sfax, Tunisia
* Corresponding author. Address: Department of General Surgery, Habib Bourguiba Hospital University of Sfax, Sfax 3021, Tunisia. Tel.: +216 2067 9500. E-mail address: drbouzidahmed89@gmail.com (A. Bouzid).
3 2023
7 2 2023
85 3 460465
8 10 2022
24 12 2022
Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc.
2023
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0/

Introduction:

Spontaneous perforation of the common bile duct (SPCBD) is an exceptional disease, especially in adults. The cause is often idiopathic once trauma and choledochal cyst are excluded. Early diagnosis is often difficult because of its often-misleading appearance.

Case presentation:

The authors report the case of a 54-year-old man, a known case of type 2 diabetes mellitus, who presented to our emergency department for acute abdominal pain that had evolved for 2 days.

Clinical findings and investigations:

On physical examination, the patient had a fever of up to 38.5°C, and the abdomen was distended and generally tight. The abdominal computerized tomography scan confirms the presence of free fluid. In addition, the thickness of the gallbladder wall had increased in contrast with a resolved gallbladder distention. Because exploration failed to demonstrate the cause of ascites, the authors performed a diagnostic paracentesis, which revealing ascites with a dark yellow-green color, which implied bile leakage.

After initial resuscitation, an emergent exploratory laparotomy was performed. The authors found biliary peritonitis secondary to SPCBD. After peritoneal lavage, the perforation was repaired over a T-tube. The postoperative course was uneventful. Histological findings were consistent with ulcerous acalculous cholecystitis. The patient was disease-free after a 3-month follow-up.

Discussion:

SPCBD is an uncommon condition in adults, and it is rarely reported in the literature. The pathogenesis of this disease is not clear and may be related to single or multiple factors. Usually, this disease is diagnosed in the postmortem. Early diagnosis and optimal surgical treatment are challenging.

Conclusion:

This present case highlighted the preoperative diagnostic difficulties of the SPCBD. Emergent surgical management should be instituted in the shortest time possible to reduce the high mortality.

Keywords:

biliary peritonitis
case report
perforation
rupture
spontaneous perforation of the common bile duct
T-tube
OPEN-ACCESSTRUE
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pmcHighlights

Often diagnosed in postmortem examination, spontaneous perforation of the common bile duct is an extremely rare condition in adults.

It is a life-threatening condition requiring an early diagnosis and emergent treatment.

Diagnostic paracentesis can help establish the preoperative diagnosis.

In most cases, the diagnosis is late and made during an explorative laparotomy.

Surgical repair over a T-tube seems to be the safest option in this situation.

Introduction

Spontaneous perforation of the common bile duct (SPCBD) is an uncommon clinical entity in which perforation occurs in the wall of the extrahepatic duct without any traumatic or iatrogenic injury. The cause is often idiopathic once trauma and choledochal cyst are excluded.

In the literature, few cases of SPCBD had been reported especially in adults and it is often diagnosed at a postmortem1. Both the diagnosis and management of this disease are ambiguous and constitute a challenge for physicians and surgeons.

Methods

This case report has been reported in line with the SCARE criteria2.

Case presentation

We present a 54-year-old man with a known case of type 2 diabetes mellitus who presented to our emergency department for acute abdominal pain evolving since two days. There is no relevant drug history, family history, genetic information, or psychosocial history related to the case. On admission, the patient had a fever of up to 38.5°C and his blood pressure was 82/45 mm Hg, with a pulse rate at 135/min. The abdomen was distended and generally tight. No jaundice was observed.

His laboratory tests revealed diabetic ketoacidosis, elevated white blood cells at 24 000/mm3, and a high C-reactive protein of 270 mg/l. The liver function tests were normal.

Abdominal ultrasonography revealed massive ascites in the abdominal cavity, especially around the liver and duodenum. The assessment of the abdominal cavity organs was difficult because of the gases filling the intestines.

The abdominal computerized tomography (CT) scan confirms the presence of free fluid. In addition, the thickness of the gallbladder wall had increased in contrast with a resolved gallbladder distention. There was no evidence of pneumoperitoneum or choledocholithiasis (Fig. 1).

Figure 1 Abdominal computerized tomography scan showing free fluid (blue arrow) associated with resolved gallbladder distention (red star).

Since noninvasive investigations failed to demonstrate the ascites’ origin, a diagnostic paracentesis was performed, revealing ascites with a dark yellow-green color, which implied bile leakage (Fig. 2).

Figure 2 Diagnostic paracentesis showing bile ascites.

After initial resuscitation, an emergent exploratory laparotomy was performed by an assistant professor in a university hospital. A diffuse biliary peritonitis was found. The majority of the bile fluid is staining the retroperitoneal cavity. After peritoneal lavage, no evidence of duodenal or gallbladder perforation was found. To explore the common bile duct, a cholecystectomy was done, and the intraoperative cholangiography showed extravasation from the posterior wall of the medium portion of the choledochus (Fig. 3). The perforation was repaired over a T-tube (Fig. 4). The peritoneal cavity was drained with two Salem tubes. The postoperative course was uneventful, and the patient was discharged on the 15th postoperative day. The procedure was successful and went as expected. The T-tube was removed on the 32 postoperative days after a normal cholangiogram excluding any biliary leakage or pancreaticobiliary junction or distal stricture (Fig. 3). The patient was satisfied and said that the symptoms had not recurred. The follow-up with the patient after 3 months remains unremarkable. Pathological examination of the cholecystectomy determined ulcerous acalculous cholecystitis.

Figure 3 Cholangiogram. (A) Perioperative findings. (B) Postoperative findings.

Figure 4 The bile duct perforation is repaired (white dotted line) over a T-tube (red star).

Discussion

Definition and etiology

SPCBD in adults is an exceptional disease. It is unrelated to traumatic or iatrogenic injury, and it is more commonly described in children due to associated congenital abnormalities in the pancreaticobiliary ductal system1,3–5. In 1882, Freeland reported the first case of SPCBD during an autopsy6.

The most frequent cause of spontaneous bile duct perforation is idiopathic. Other causes were reported in the literature, such as common bile duct stones, previous biliary tract surgeries, tumors, congenital malformations like choledochal cysts, and biliary diverticulum1,3.

The pathogenesis of spontaneous perforation of the bile duct has not been clarified and may be related to single or multiple factors. It has been suggested that congenital weakness or ischemia of the bile duct wall, a sudden increase in the bile duct pressure such as biliary stones, and pancreaticobiliary reflux can lead to perforation of the bile duct1,5,7.

Diagnosis

The diagnosis of SPCBD is difficult to establish before surgery. Both clinical and laboratory findings are variable and unspecific. Patients may present with a variable spectrum of disease severity, ranging from a localized right upper quadrant inflammatory process to diffuse peritonitis.

In the literature, ultrasonography and CT scans can show indirect signs of perforation such as gallbladder decompression, perihepatic collection, or free fluid in the abdominal cavity. However, the exact site of the perforation remains difficult to predict. In a few cases, the preoperative diagnosis was established by an iminodiacetic acid scan, magnetic resonance cholangiopancreatography, endoscopic retrograde cholangiopancreatography, MRI, and CT scan8,9.

Moreover, some authors recommend a diagnostic paracentesis when doubt exists for perforation of the biliary tree (failure to demonstrate the gallbladder, predominant ascites within a lesser sac or in the right quadrant, the presence of a deviated calculus from the bile duct, etc.)3,8,10–16Despite the advances in imaging tools and intensive care management, delayed diagnosis still results in high morbidity and mortality1,3,7,10.

Treatment

The surgical management of the biliary duct perforation remains controversial, but the goals of this surgical treatment are to treat abdominal contamination with infected bile and close the biliary perforation.

When an extraductal obstruction is identified, surgery often offers the advantage of treating the disease and the complication.

To our knowledge, only 14 cases of SPCBD in adults were reported from 2010 to 2022. The clinical characteristics and surgical outcomes are mentioned in Table 1 3,8–15,18–22.

Table 1 Clinical features and surgical outcomes of 15 case reports (including our case) of spontaneous common bile duct perforation in adults searched from 2010 to 2022

References	Age	Sex	Imaging modality for preoperative diagnosis	Paracentesis	Preoperative diagnosis	Causes	Surgical procedure (emergency)	Location of perforation	Outcome	
Khanna10 2010	50	F	ND	Biliary fluid	Peptic perforation	Stone	T-tube drainage	Supraduodenal	ND	
Laway11 2013	35	M	US, CT	Biliary fluid	Duodenal perforation	ND	T-tube drainage, cholecystectomy	Supraduodenal	Alive	
Paramhans18 2013	44	F	US	Not performed	N.D	Infection	T-tube drainage, cholecystectomy	CBD at cystic junction	Alive	
Bowan19 2013	29	F	ND	Biliary fluid	Peptic perforation	Stone, pregnancy	T-tube drainage, cholecystectomy	2 parts: hepatic duct and supraduodenal	Alive	
Singh20 2014	25	F	US	Not performed	ND	Choledochal cyst, pancreatitis, pregnancy	T-tube drainage, cholecystectomy	2 parts: mid and supraduodenal	Alive	
Ishii12 2016	82	M	US, CT	Not performed	Hemorrhage/colitis/appendicitis	Stone	RTBD, cholecystectomy	Left hepatic duct	Alive	
Pulat9 2016	36	F	US, CT	Not performed	Perforation of CBD	Pancreatitis	T-tube drainage, cholecystectomy	Common hepatic duct	Alive	
Subasinghe13 2016	66	F	US	Not performed	Peritonitis	Stone	T-tube drainage, cholecystectomy	CBD at cystic junction	Alive	
Hamura3 2016	84	M	US, CT, (postoperative ERCP)	Biliary fluid	Bile peritonitis	Stone	Abdominal lavage only	ND	Dead	
Mohanty21 2017	17	F	US	Biliary fluid	Peptic perforation	ND	T-tube drainage, cholecystectomy	Supraduodenal	Alive	
Huda22 2017	40	M	US	Biliary fluid	Perforation of CBD	ND	T-tube drainage, cholecystectomy	Supraduodenal	Alive	
Amberger8 2018	28	F	US, CT, MRI, Scintigraphy	Biliary fluid	Perforation of CBD	ND	(ERCP case)	Supraduodenal	Alive	
Sakamoto14 2021	71	F	US, CT	Biliary fluid	Perforation of CBD	Maljunction, stone, pancreatitis, cholangitis	T-tube drainage, cholecystectomy	Supraduodenal	Alive	
Gkionis15 2022	89	M	US, CT	Not performed	Peritonitis	ND	Not completed	ND	Dead	
This study	54	M	US, CT	Biliary fluid	Bile peritonitis	ND	T-tube drainage, cholecystectomy	Supraduodenal	Alive	
CBD, common bile duct; CT, computed tomography; ERBD, endoscopic retrograde biliary drainage; ERCP, endoscopic retrograde cholangiopancreatography; ND, not described; US, ultrasonography.

The optimal management of common duct bile perforation is cholecystectomy associated with intraoperative cholangiography via a transcystic stent. Traditionally, the perforation is closed over a T-tube1,17,23. In absence of biliary stenosis or pancreaticobiliary maljunction, primary closure has been suggested to be as safe as a closure over a T-tube. In these optimal conditions, cholecystectomy and peritoneal irrigation with perihepatic drainage can be another therapeutic option. The perforation will be filled spontaneously without T-tube drainage.

With the recent advancements in stenting and lessons learned from iatrogenic lesions, conservative treatment including biliary stenting by endoscopic retrograde cholangiopancreatography associated with percutaneous drainage should be considered and can be an alternative to surgery in stable patients and cases of local peritonitis16.

In all reported cases, the surgical approach for spontaneous bile duct perforation was a laparotomy to ensure complete peritoneal lavage and bile duct repair. However, in certain instances (absence of septic shock, preoperative diagnosis of local peritonitis), it seems that a minimally invasive approach through laparoscopy can be a safe option.

In our case, the closure was over a T-tube to perform a postoperative cholangiogram and diagnose any leak into the peritoneal cavity10–15,17–23. Also, we remember that we have been dealing with diffuse peritonitis in unstable patients and have preferred to perform an open surgical approach.

Conclusion

Spontaneous perforation of the extrahepatic bile duct is a rare condition in adults that needs an early diagnosis. Emergent surgical management should be instituted in the shortest time possible to reduce the high mortality.

Ethical approval

No ethical approval was necessary.

Consent

Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Sources of funding

The author(s) received no financial support for this article’s research, authorship, and/or publication.

Author contribution

A.B. conceived and designed the study. A.B., S.F., and H.R. were responsible for data collection and acquisition of data. A.B., S.F., and A.T. analyzed and/or interpreted the data. Y.M. and S.F. performed the literature review. A.B., S.F., and Y.M. wrote the initial manuscript. H.R. and S.B. critically revised the manuscript. All authors have approved the final manuscript.

Conflicts of interest disclosure

No conflict of interest.

Research registration unique identifying number (UIN)

Not available.

Guarantor

Ahmed Bouzid.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Sponsorships or competing interests that may be relevant to content are disclosed at the end of this article.

Published online 7 February 2023
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