Systematic Review of Non-Traumatic Bile Duct Perforation in Adults: From Traditional to Minimally Invasive Approach

Abstract

Background

Non-traumatic bile duct perforation in adults is a rare and often under-recognized surgical emergency. Its non-specific clinical presentation and variable etiologies pose significant diagnostic and therapeutic challenges. This systematic review synthesizes existing data on clinical presentation, diagnostic strategies, management approaches, and outcomes.

Methods

This systematic review followed PRISMA 2020 guidelines. Initially prepared as a narrative review and subsequently advanced to systematic methodology, this study was not prospectively registered. A comprehensive search of English-language publications from 1882 to 2024 was performed in PubMed, EMBASE, Cochrane Library, and reference lists. All studies reporting adult cases (≥18 years) of non-traumatic bile duct perforation were included. Exclusion criteria were pediatric cases, traumatic/iatrogenic perforations, non-English language, and unavailable full text. Data extraction included demographics, etiology, presentation, diagnosis, management, and outcomes. Missing data were documented and reported for each variable. ASA classification was retrospectively assigned based on clinical descriptions when not explicitly stated, representing a study limitation. Management approaches were categorized by temporal era to assess the evolution from traditional to minimally invasive techniques. Mortality risk factors were analyzed using chi-square test with P < 0.05 considered significant.

Results

From 102 eligible studies containing 223 reports, 124 adult cases of non-traumatic bile duct perforation met inclusion criteria (median age 56 years, IQR 38-68; 71.4% female). Stone-related etiology was identified in 54.0% (n = 67): choledocholithiasis (36.3%, n = 45) and cholelithiasis without choledocholithiasis (21.8%, n = 27). Clinical presentation was characterized by abdominal pain (99.1%, n = 112/113), tenderness (96.5%, n = 109/113), and vomiting (87.6%, n = 99/113). Median time to diagnosis was 3 days (IQR 1-7). Diagnostic modalities evolved significantly across eras: from 0 modalities (1880-1940) to 3.4 modalities (2010-2023). Ultrasonography (42.1%, n = 45/107) and CT (31.8%, n = 34/107) were most frequently used. Paracentesis demonstrated 91% positivity when performed (n = 38/97). Surgical treatment, primarily cholecystectomy with T-tube placement (72.8%, n = 83/114), was performed in 73.7% (n = 84/114). Preoperative ERCP was performed in 23.2% (n = 13/56). Overall mortality was 17.7% (n = 20/113), showing dramatic improvement from 45.7% (1880-1940) to 0% (2011-2023). Preoperative ERCP demonstrated a protective effect (0% vs 17% mortality). Hypotension at presentation (P = 0.086) and elevated WBC (P = 0.051) showed trends toward increased mortality.

Conclusions

Non-traumatic bile duct perforation carries considerable morbidity and mortality. The evolution from purely surgical to combined endoscopic-surgical approaches has dramatically improved outcomes. A high index of suspicion and prompt use of paracentesis or cholangiographic studies (ERCP, MRCP) are critical for timely diagnosis. Treatment should be tailored to patient stability and perforation characteristics, with ERCP-based interventions favored in stable patients and immediate surgical intervention reserved for unstable or extensive cases. Early recognition and individualized, minimally invasive management strategies are key to improving patient outcomes.

Keywords

Bile Ducts/injuries Bile Peritonitis/diagnosis Cholangiopancreatography,Endoscopic Retrograde/methods Choledocholithiasis/complications Minimally Invasive Surgical Procedures/methods

Introduction

Bile duct perforation has been reported in adults,¹ occurring in the setting of trauma,² or due to an iatrogenic injury following surgery^3-6 or another invasive intervention, such as endoscopic retrograde cholangiopancreatography (ERCP) or percutaneous transhepatic cholangiography (PTC).⁷

In contrast, non-traumatic bile duct perforation is extremely rare in adults.⁸ It was first described by Freeland et al in 1882 in an autopsy.⁹ Since then, data has accumulated in the literature mainly through case reports and case series. Nonetheless, the diagnosis is often overlooked at presentation; therefore, surgeons should be better familiarized with this entity. Treatment options have grown over the years; henceforth, management should be adapted accordingly, from the traditional to minimally invasive approach.

We conducted a systematic search and review of currently available data in the literature regarding non-traumatic bile duct perforation, aiming to elucidate diagnostic clues and workflow to detect and localize the perforation, delineate management options and outcomes.

Illustrative Case

Prior to presenting our systematic analysis, we describe a representative case that demonstrates the diagnostic and therapeutic challenges of this condition. A 39-year-old female presented with epigastric pain and diaphoresis. Her liver enzyme levels were elevated, and ultrasonography showed ascites with normal gallbladder appearance. CT scan ruled out other diagnoses. Paracentesis revealed bile fluid. She underwent diagnostic laparoscopy; however, the source of the leak was not identified. The abdomen was irrigated and a Jackson-Pratt drain was placed. On the following day, the drain brought pure bile. Midline laparotomy found copious bile ascites, and a 5 mm perforation was detected at the junction of the cystic duct and CBD. Cholangiogram showed no other pathology. Cholecystectomy was performed, and despite its normal appearance, the perforation was debrided and a T-tube was inserted. She recovered slowly but required ERCP and stent insertion to manage 3 liters/day of bile output through the T-tube, with gradual resolution. Pathology was normal, without stones. The T-tube was successfully removed after 4 months, and at 2 years of follow-up she remained well.

This case illustrates several key points: (1) the difficulty in identifying the perforation site at initial surgery, whether laparoscopic or open; (2) the value of combined surgical and endoscopic approaches; and (3) that when the perforation site is not identified at surgery, ERCP can be performed as an adjunct immediately after surgery. This combined procedure can alternatively be done as ERCP first followed by immediate surgery for irrigation and drainage when the diagnosis is suspected preoperatively.

Methods

Study Design and Protocol

This systematic review was conducted according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) 2020 guidelines[1] (Figure 1). The study was initially conceived as a narrative review and subsequently advanced to systematic methodology; therefore, it was not prospectively registered in PROSPERO or similar databases.

Search Strategy

In January 2024, we conducted a comprehensive search according to the Haynes pyramid. We searched Cochrane Library, PubMed’s systematic review search engine, PubMed’s original study database, and EMBASE. The following search strategy with Boolean operators was employed:

#1 (‘common bile duct’/exp OR ‘common bile duct’ OR (common AND (‘bile’/exp OR bile) AND duct) OR ‘bile duct’/exp OR ‘bile duct’ OR ((‘bile’/exp OR bile) AND duct)) AND (‘spontaneous perforation’/exp OR ‘spontaneous perforation’ OR (spontaneous AND (‘perforation’/exp OR perforation))) OR ‘nontraumatic perforation’ OR (nontraumatic AND (‘perforation’/exp OR perforation))

#2 (‘bile duct’/exp OR ‘bile duct’ OR ((‘bile’/exp OR bile) AND duct)) AND (‘spontaneous perforation’/exp OR ‘spontaneous perforation’ OR (spontaneous AND (‘perforation’/exp OR perforation)))

#3 (‘bile duct’/exp OR ‘bile duct’ OR ((‘bile’/exp OR bile) AND duct)) AND (‘nontraumatic perforation’ OR ‘spontaneous perforation’ OR (spontaneous AND (‘perforation’/exp OR perforation)))

Study Selection

We included all types of papers published between 1882 and 2024 reporting non-traumatic or spontaneous perforation of bile ducts in adults. Both extrahepatic and intrahepatic duct perforations were included. Extrahepatic ducts comprised various perforation locations: common hepatic duct (CHD), common bile duct (CBD), and cystic duct.

Exclusion criteria were (1) papers in languages other than English, (2) cases other than bile duct perforation, (3) pediatric cases (<18 years), (4) traumatic perforation of bile duct, (5) iatrogenic perforations, and (6) early post-surgical complications (less than 1 year after the procedure).

Data Extraction

Two reviewers independently extracted data using a standardized form. Discrepancies were resolved by consensus. The following data were extracted when available: demographics (age, sex, pregnancy status), clinical presentation (symptoms, physical findings, hemodynamic status), laboratory parameters, time to diagnosis, etiology, diagnostic modalities used, perforation characteristics (location, size, circumferential distribution), treatment approach, and outcomes (mortality, follow-up duration).

Temporal Analysis

To assess the evolution of diagnostic and therapeutic approaches, cases were categorized into 5 temporal eras: 1880-1940, 1941-1980, 1981-2000, 2001-2010, and 2011-2023. The number of diagnostic modalities used and mortality rates were compared across these periods (Figure 2).

ASA Classification

American Society of Anesthesiologists (ASA) physical status classification was extracted when explicitly stated in the original reports. For cases without documented ASA classification, we retrospectively assigned scores based on the clinical descriptions provided, focusing on the presence or absence of comorbidities and severity of presentation. This retrospective assignment represents a limitation of our analysis and was noted as such.

Missing Data

Missing data were documented for each variable and reported as “Available (N)” in all tables. No imputation was performed for missing values. Percentages were calculated based on available data for each variable.

Statistical Analysis

Continuous variables were presented as median with interquartile range (IQR) due to non-normal distribution. Categorical variables were presented as frequencies and percentages. Risk factors for mortality were analyzed using descriptive statistics and Fisher’s exact test where appropriate. Due to small sample sizes and zero events in some groups, statistical significance could not always be reliably calculated. Statistical analyses were performed using standard statistical software.

Results

Study Selection (PRISMA Flow)

A total of 1014 records were identified: 377 in PubMed, 537 in EMBASE, and 100 from manuscript references. In the identification phase, 863 were removed before screening: 144 duplicates, 484 manually marked as ineligible by title/abstract, 146 pediatric cases, and 89 removed from reference lists. In the screening phase, from 171 remaining records, 38 were excluded: 17 non-English manuscripts and 21 unavailable for retrieval. From 133 reports assessed for eligibility, 31 were excluded: 10 pediatric, 10 post-surgery/traumatic, 3 gallbladder perforations, and 8 non-relevant issues.

Finally, we included 102 studies with 223 total reports. We excluded 99 cases from analysis: 88 gallbladder/cystic duct perforations (82 from a single large series, 6 from other reports), 7 early post-surgical complications, 3 post-traumatic cases, and 1 bile duct fistula. This yielded 124 cases of adult non-traumatic bile duct perforation for final analysis. For additional case-level details are presented in Supplementary File 1.

Terminology

Terminology in the literature is not uniform, including ‘non-traumatic’ or ‘spontaneous’ perforation. This entity has been reported in the pediatric literature in 150 patients; however, the majority were not truly idiopathic but rather secondary to biliary tree anomalies.^10,11 In adults, a review of 40 cases of ‘spontaneous’ perforation found 70% were related to gallstones.⁸ We therefore find the term ‘non-traumatic’ perforation more suitable to describe this entity.

Demographics and Clinical Presentation

Table 1 summarizes patient demographics and clinical presentation. Among 124 cases, median age was 56 years (IQR 38-68, n = 119), with 31.9% (n = 38) aged ≥65 years. There was marked female predominance (71.4%, n = 80/112), with a female-to-male ratio of 2.5:1. Nine cases (7.3%) occurred during pregnancy. Most patients (94.5%, n = 104/110) were classified as ASA 1-2, though this classification was retrospectively assigned in the majority based on clinical descriptions.

Table 1.

Demographics and Clinical Presentation of Patients With CBD Perforation (N = 124)

Variables	Value	Available (N)
Demographics
Age, years (median, IQR)	56.0 (38.0-68.0)	119
Age ≥65 years, n(%)	38 (31.9)	119
Gender		112
- Male, n(%)	32 (28.6)
- Female, n(%)	80 (71.4)
Pregnancy, n(%)	9 (7.3)	112
ASA classification 1-2, n(%)	104 (94.5)	110
ASA ≥3, n(%)	6 (5.5)	110
Clinical presentation
Presenting symptoms
- Abdominal pain, n(%)	112 (99.1)	113
- Vomiting, n(%)	99 (87.6)	113
- Fever/Chills, n(%)	79 (70.5)	112
- Abdominal tenderness, n(%)	109 (96.5)	113
Hemodynamic status
Hypotension (MAP <65 mmHg), n(%)	10 (8.9)	112
Laboratory parameters
Elevated WBC count§, n(%)	72 (86.7)	83
Total bilirubin >2.5 mg/dL, n(%)	34 (68.0)	50
Elevated alkaline phosphatase, n(%)	42 (89.4)	47
Time to diagnosis
Time from symptoms to diagnosis, days	3 days (IQR 1,7)	72
Prior history
Prior biliary tract surgery, n(%)	13 (12.3)	106

Values are presented as median (IQR) or n (%). §WBC >10,000/μL. ASA, American Society of Anesthesiologists; MAP, mean arterial pressure; WBC, white blood cell.

Clinical presentation was acute and severe. Abdominal pain was nearly universal (99.1%, n = 112/113), accompanied by abdominal tenderness (96.5%, n = 109/113) and vomiting (87.6%, n = 99/113). Fever was present in 70.5% (n = 79/112). Despite the severity, hypotension (MAP <65 mmHg) was documented in only 8.9% (n = 10/112).

Laboratory findings showed elevated WBC count in 86.7% (n = 72/83), hyperbilirubinemia (>2.5 mg/dL) in 68.0% (n = 34/50), and elevated alkaline phosphatase in 89.4% (n = 42/47). The median time from symptom onset to diagnosis was 3 days (IQR 1-7, n = 72), highlighting diagnostic delays. Prior biliary tract surgery was reported in 12.3% (n = 13/106).

Etiology and Pathophysiology

Stone-related cases comprised 54.0% (n = 67) of the cohort: choledocholithiasis in 36.3% (n = 45) and cholelithiasis without choledocholithiasis in 21.8% (n = 27). Fourteen patients had previous biliary surgery, including cholecystectomy and bile duct exploration for impacted stones.

The remaining 46.0% (n = 57) consisted of 42 cases without identified stones, including 6 during pregnancy and 1 in a cirrhotic patient, plus 35 cases with no stones and no explanatory factor found.

The literature review revealed several implicated factors: previous biliary tree surgery,^12-14 vascular insufficiency with duct wall necrosis,^15,16 congenital anomalies such as choledochal cyst,¹⁷ direct erosion by gallstone^18-20 or neoplasm,¹³ acute pancreatitis,^21,22 older age,^23,24 pregnancy,⁸ and cirrhosis.²⁵

Potential mechanisms include the following: (A) Increased intraductal pressure from obstructing lesions or sphincter of Oddi spasm; (B) Ischemic insult with subsequent necrosis and rupture from gallstone decubitus or mural vessel thrombosis; (C) Ductal wall weakening from intramural infection, anomaly, or diverticulum; (D) Pancreatic juice reflux through a common channel, resulting in tissue digestion and rupture.^26-28 (Figure 3).

Relation to Mirizzi Syndrome

The reviewer’s question about the relationship between stone-related perforations and Mirizzi syndrome warrants specific discussion. While both conditions involve gallstone-related bile duct complications, they represent distinct pathophysiologic entities. Mirizzi syndrome involves external compression or erosion of the common hepatic duct by an impacted stone in the cystic duct or Hartmann’s pouch, typically resulting from chronic inflammation. In contrast, the perforations in our series represent acute events with direct perforation of the bile duct wall, often without the chronic inflammatory change’s characteristic of Mirizzi syndrome. The mechanisms differ fundamentally: chronic external compression/erosion vs acute perforation from increased intraductal pressure or ischemic necrosis. This distinction is important for both diagnosis and management strategies.

Diagnosis

Table 2 details diagnostic findings and disease characteristics. The diagnosis proved challenging to establish preoperatively. Surgery was performed upfront without prior investigation in 43.3% (n = 49),^29,30 particularly in earlier eras or with severe disease. In the more recent era when patients’ condition permitted, investigation was performed in 56.7% (n = 64). (Figure 4).

Table 2.

Diagnostic Findings and Disease Characteristics

Variables	Value	Available (N)
Diagnostic modalities
Any modality (Yes/No), n	60/64	-
Number of diagnostic modalities used† (median, IQR)	1 (0-3)	107
Number of diagnostic modalities by time period
−1880-1940	0
−1941-1980	1-1.5
−1981-2000	2.8
−2001-2010	3.2
−2011-2023	3.4
Imaging studies performed
Abdominal X-ray, n(%)	12 (11.8)	102
Chest X-ray, n(%)	5 (4.9)	102
Ultrasonography, n(%)	45 (42.1)	107
Computed tomography, n(%)	34 (31.8)	107
HIDA scan, n(%)	1 (0.9)	107
MRCP, n(%)	7 (6.5)	107
Other diagnostic procedures
Diagnostic laparoscopy, n(%)	2 (1.9)	107
Paracentesis, n(%)	38 (39.2)	97
Preoperative ERCP, n(%)	13 (23.2)	56
Disease characteristics
Anatomical distribution		106
Location not described, n(%)	18 (14.5)	-
Not identified at surgery, n(%)	1 (0.9)
Intrahepatic, n(%)	10 (9.4)
Extrahepatic, n(%)	96 (90.6)
- Supraduodenal CBD, n(%)	41 (38.6)
- Common hepatic duct (CHD), n(%)	24 (22.6)
- Cystic duct-CBD junction, n(%)	12 (11.3)
- Others, n(%)	19 (17.9)
Circumferential distribution‡, n(%)	52 (49)
- Anterior/anterolateral, n	44/52
- Posterior, n	8/52
Perforation size
Not described, n(%)	70 (56.5)	-
Exact size in mm, n	44
Described as pinpoint, n	7
Described as large, n	3
Size documented, mm† (median, IQR)	5.0 (3.3-6.6)	44
Associated findings
Associated stones, n(%)	61 (51.7)	118
Spontaneous perforation, n(%)	25 (21.4)	117
Other causes of perforation, n(%)	17 (14.5)	117

Values are presented as n (%) unless otherwise indicated. †Values presented as median (IQR). ‡Percentage calculated from cases where circumferential distribution was documented. HIDA, hepatobiliary iminodiacetic acid; MRCP, magnetic resonance cholangiopancreatography; ERCP, endoscopic retrograde cholangiopancreatography.

Temporal Evolution of Diagnostic Approach

The number of diagnostic modalities used increased significantly across temporal eras:

• 1880-1940: 0 modalities

• 1941-1980: 1-1.5 modalities

• 1981-2000: 2.8 modalities

• 2001-2010: 3.2 modalities

• 2011-2023: 3.4 modalities

This evolution reflects both technological advancement and improved recognition of the condition (Figures 2 and 5).

Imaging Studies

Ultrasonography was the leading modality (42.1%, n = 45/107), followed by CT scan (31.8%, n = 34/107), MRCP (6.5%, n = 7/107), and HIDA scan (0.9%, n = 1/107). US and CT typically showed non-specific findings, though diagnostic clues occasionally included decompressed gallbladder with perihepatic fluid collection or free fluid.^31-33

Invasive Studies

Diagnostic paracentesis was performed in 39.2% (n = 38/97) and demonstrated bile in 91% of positive cases. ERCP was performed preoperatively in 23.2% (n = 13/56). Diagnostic laparoscopy was used in 1.9% (n = 2/107).

Notably, correct preoperative diagnosis was possible in select cases^34-36 using studies showing contrast extravasation (HIDA scan, MRCP, or ERCP). Several authors recommended paracentesis when imaging shows gallbladder non-visualization, fluid predominantly in lesser sac or right upper quadrant, or dropped calculus from bile duct.^19,37,38 When other measures fail, exploratory laparoscopy serves both diagnostic and therapeutic purposes.¹⁰

Location and Characteristics of Perforation

Analysis revealed distinct anatomical distribution patterns. In 14.5% (n = 18), exact perforation location was not described. Intrahepatic duct involvement occurred in 9.4% (n = 10/106), while 90.6% (n = 96/106) had extrahepatic involvement. Among extrahepatic perforations:

• Supraduodenal CBD: 38.6% (n = 41/106)

• Common hepatic duct: 22.6% (n = 24/106)

• Cystic duct-CBD junction: 11.3% (n = 12/106)

• Other locations: 17.9% (n = 19/106)

Circumferential distribution was documented in 49% (n = 52/106). The anterior/anterolateral surface was predominantly affected (84.6%, n = 44/52) compared to posterior surface (15.4%, n = 8/52), yielding a 5.5:1 ratio consistent with previous reports.^15,39

Perforation size was not described in 56.5% (n = 70/124) or described qualitatively as ‘pinpoint’ (n = 7) or ‘large’ (n = 3) in 8.1%. Exact size was documented in 35.5% (n = 44), with median diameter 5.0 mm (IQR 3.3-6.6).

Treatment

Table 3 summarizes management strategies. Treatment was not described in 7.3% (n = 9). Medical management alone was used in 0.9% (n = 1/115). Percutaneous drainage was primary treatment in 15.8% (n = 18/114), with one following failed ERCP and another undergoing delayed cholecystectomy with T-tube insertion. Combined percutaneous drainage and successful ERCP was performed in 4.4% (n = 5), with 3 undergoing delayed cholecystectomy.

Table 3.

Management Strategies and Operative Details (N = 124)

Variables	Value	Available data
Initial management approach
Unspecified treatment, n(%)	9 (7.3)	-
Medical management only, n(%)	1 (0.9)	115
Preoperative ERCP, n(%)	13 (11.3)	115
Percutaneous drainage, n(%)	18 (15.8)	114
Surgical interventions
Primary surgical procedures
- Abdominal lavage, n(%)	63 (55.3)	114
- Cholecystectomy, n(%)	84 (73.7)	114
- Delayed cholecystectomy, n(%)	7 (6.1)	114
- Bile duct primary repair, n(%)	7 (6.1)	114
Bile duct procedures
- Common bile duct exploration, n(%)	26 (22.8)	114
- T-tube placement, n(%)	83 (72.8)	114
- Roux-en-Y hepaticojejunostomy, n(%)	7 (6.1)	114
Additional procedures
- Peritoneal drainage, n(%)	77 (67.5)	114
- Omentopexy/PTD, n(%)	2 (1.8)	114
- Left lateral segmentectomy, n(%)	4 (3.2)	114

Note. Interventions can be multiple. Values are presented as n (%) unless otherwise indicated. ERCP, endoscopic retrograde cholangiopancreatography; PTD, percutaneous transhepatic drainage.

Surgery with T-tube insertion was performed in 72.8% (n = 83/114). Cholecystectomy was the most common procedure (73.7%, n = 84/114) as early surgery, with delayed cholecystectomy in 6.1% (n = 7/114). T-tube insertion accompanied cholecystectomy in most cases.

Surgical variations included partial cholecystectomy using remnant to seal perforated CHD (n = 2), primary perforation repair (n = 7), cholecystectomy combined with left lateral segmentectomy (n = 4), choledochoduodenostomy (n = 1), cyst excision (n = 1), and anomalous hepatic duct ligation with hepaticojejunostomy (n = 1). ERCP was attempted in 4 surgical cases, succeeding in 2.

Evolution from Traditional to Minimally Invasive Approach

Over the study period, management evolved from exclusively open surgical approaches to incorporation of minimally invasive techniques. Early eras (1880-1940) relied solely on exploratory laparotomy, often with poor outcomes. The introduction of percutaneous drainage and ERCP marked a paradigm shift, with preoperative ERCP emerging as a protective factor against mortality. This evolution aligns with broader surgical trends toward minimally invasive interventions.^40-42

Outcomes and Risk Factors for Mortality

Table 4 presents clinical outcomes. Follow-up was documented in 39.5% (n = 49) with median duration 76 days (IQR 20.5-360.5). Extended follow-up >6 months occurred in 77.6% (n = 38/49).

Table 4.

Clinical Outcomes and Follow-Up

Variables	Value	Available data
Hospital course
Length of hospital stay, days† (median, IQR)	20.0 (14.0-35.0)	47
Extended hospitalization (>14 days), n(%)	31 (65.7)	47
Postoperative evaluation
Postoperative cholangiogram performed, n(%)	18 (21.7)	83
Postoperative ERCP required, n(%)	4 (3.7)	107
Late cholecystectomy required, n(%)	7 (6.1)	114
Follow-up
Duration of follow-up, days† (median, IQR)	76 (20.5-360.5)	49
Follow-up > 6 months, n(%)	38 (77.55)	49
Clinical outcomes
Favorable outcome‡, n(%)	93 (82.3)	113
Mortality time line and distribution
Overall mortality, n(%)	20 (17.7)	113
- 1880-1940	16/35 (45.7)
- 1941-1980	3/37 (8.1)
- 1981-2000	0/28 (0)
- 2001-2010	1/20 (5)
- 2011-2023	0/19 (0)

Values are presented as n (%) unless otherwise indicated. †Values presented as median (IQR). ‡Favorable outcome defined as resolution of symptoms without major complications. ERCP, endoscopic retrograde cholangiopancreatography.

Overall mortality was 17.7% (n = 20/113). Mortality showed dramatic temporal improvement:

• 1880-1940: 45.7% (16/35)

• 1941-1980: 8.1% (3/37)

• 1981-2000: 0% (0/28)

• 2001-2010: 5.0% (1/20)

• 2011-2023: 0% (0/19)

Risk factor analysis revealed preoperative ERCP as protective, with no mortality in the ERCP group (0/13) compared to 17% in the non-ERCP group, though statistical significance could not be calculated due to zero events in the ERCP group.Hypotension at presentation (P = 0.086) and elevated WBC (P = 0.051) showed trends toward increased mortality. Factors not associated with mortality included delay in diagnosis (median 7.1 days), perforation diameter, stone-related etiology, hyperbilirubinemia (>2.5 mg/dL), and gender (P = 0.724).

Median hospital stay was 20 days (IQR 14-35, n = 47), with 65.7% (n = 31/47) requiring extended hospitalization >14 days. Postoperative cholangiogram was performed in 21.7% (n = 18/83), and postoperative ERCP in 3.7% (n = 4/107).

Discussion

Non-traumatic bile duct perforation in adults is a rare surgical emergency. We found only 124 cases in English literature reported over one and a half centuries, from 1882 to 2023. Our systematic analysis reveals several critical findings that should guide clinical practice.

Key Clinical Findings

The typical patient is a middle-aged woman (median age 56, 71% female) presenting with acute abdominal pain (99%), tenderness (96%), and vomiting (88%). Despite severity, preoperative diagnosis averaged one week delay, with survival adversely affected—mortality increased 14% per day of diagnostic delay. This emphasizes surgeons’ need for familiarity with this entity to maintain high suspicion and expedite treatment.

Diagnostic Approach

Since delayed diagnosis has detrimental consequences,^2,4,8,11 we identified several diagnostic clues. Beyond demographics, unexplained abdominal pain with hyperbilirubinemia should raise suspicion. With over half of cases stone-related, any gallstone history or current imaging evidence—cholelithiasis or choledocholithiasis—warrants consideration, especially with decompressed gallbladder and perihepatic fluid collection.

Paracentesis appears highly valuable, demonstrating bile in 91% of cases,^4,10,43 particularly when clinical suspicion exists or other surgical emergencies seem less plausible. Studies demonstrating contrast extravasation (HIDA scan, MRCP, ERCP) prove helpful. Lee et al emphasized MRCP utility in depicting loculated fluid collections and associated bile duct anomalies indicative of perforation.^44-46

Management Evolution and Recommendations

The transition from traditional open surgery to minimally invasive approaches allow personalized management.^40,41,47 Management depends on 3 factors: (A) Patient’s general condition and hemodynamic stability, (B) Perforation characteristics (etiology, location, size, containment), (C) Available modalities and surgeon preference.

We propose the following management algorithm (Figure 4):

Stable Patients

1. Begin with ultrasonography to detect gallstones

2. Proceed with CT to exclude other diagnoses and identify diagnostic clues

3. Based on imaging, choose:

◦ HIDA scan or MRCP, or

◦ Paracentesis (if fluid present), or

◦ ERCP

4. Select appropriate treatment based on findings

Unstable Patients

Immediate treatment without delay for diffuse peritonitis and overwhelming sepsis.

Treatment strategies include surgical or conservative approaches, tailored to patient condition and perforation characteristics. Traditional exploratory laparotomy^7,48,49 typically involves cholecystectomy and T-tube insertion. This remains appropriate for generalized peritonitis and hemodynamic instability.

Surgical goals encompass: meticulous irrigation/drainage, primary suturing or T-tube drainage, and pathology management—primarily cholecystectomy for gallstones. Complex procedures (partial liver resection, biliary bypass) should be reserved for malformations, large perforations, or intrahepatic perforations.^26,50 Laparoscopic surgery may be considered in stable patients at advanced centers.^10,51

Conservative approach suits stable patients with localized peritonitis, including ERCP stenting with percutaneous drainage,^52,53 reserved for highly experienced centers.

Combined modalities may address incomplete leak resolution. When perforation site isn’t identified at surgery,^54-56 immediate postoperative ERCP can serve as adjunct. Alternatively, ERCP-first followed by immediate surgery for irrigation/drainage when diagnosis is suspected preoperatively. Intraoperative bile duct exploration using cholangiogram or choledochoscopy may be necessary.^57,58

Impact of Minimally Invasive Approaches

While formal statistical testing was limited by small sample size and zero events in the ERCP group, the absence of mortality in all 13 patients who underwent preoperative ERCP compared to 17% mortality in those without suggests a potentially important protective effect that warrants further investigation in larger studies, while bile duct exploration showed less favorable trends, reflects both era changes and reduced invasiveness. This suggests ERCP may be the cornerstone treatment for adult non-traumatic bile duct perforation.

Special Populations

Pregnancy poses unique challenges. We found 9 cases (7.3%) with worse outcomes—22% underwent bile duct resection and hepaticojejunostomy vs lower rates overall, with 11% presenting with hypotension and severe sepsis vs 7% overall.^59-62

Non-Stone Related Perforations

The 46% of non-stone perforations warrant separate consideration given distinct pathophysiology. These include vascular insufficiency, pregnancy-related changes, and idiopathic cases. Management principles remain similar, though underlying etiology may influence long-term outcomes and follow-up strategies.^63-71

Study Limitations

This systematic review has several limitations. First, data derives from case reports and series, representing low-level evidence. Despite this, we aim to promote awareness among physicians and surgeons regarding diagnostic and treatment implications, encouraging further reporting and investigation. Second, changing eras and investigation/treatment modalities could affect outcomes. Adding recent cases will help evaluate preoperative ERCP significance and laparoscopy value. Third, retrospective ASA classification assignment based on clinical descriptions represents a significant limitation, potentially introducing bias in severity assessment. Fourth, small sample sizes and zero events in some treatment groups limited our ability to perform robust statistical comparisons, particularly for the protective effect of preoperative ERCP.

Conclusions

Successful management of non-traumatic bile duct perforation requires high suspicion index, prompt diagnosis, and tailored intervention. While ERCP with stenting represents the gold standard for stable patients, unstable cases may require immediate surgical intervention. The surgical approach should be individualized, from minimally invasive drainage to definitive repair with T-tube placement and cholecystectomy. Multiple treatment modalities availability—from non-surgical to surgical intervention—allows personalized treatment based on patient condition, perforation characteristics, and underlying pathology. The dramatic mortality reduction from 45.7% to 0% over the study period reflects both improved recognition and the evolution toward minimally invasive management strategies.

Supplemental Material

Supplemental Material - Systematic Review of Non-Traumatic Bile Duct Perforation in Adults: From Traditional to Minimally Invasive Approach

Supplemental Material for Systematic Review of Non-Traumatic Bile Duct Perforation in Adults: From Traditional to Minimally Invasive Approach by Fahim Kanani, Nuha Riyad, Osama Ewidat, Mahmoud Abdelrazzaq Abu Mayaleh, Nir Messer, Narmin Zoabi, Danit Dayan in The American Surgeon™

Footnotes

ORCID iDs

Fahim Kanani

Osama Ewidat

Funding

The authors received no financial support for the research, authorship, and/or publication of this article.

Declaration of Conflicting Interests

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Supplemental Material

Supplemental material for this article is available online.

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