A rare case of obstructive shock caused by a giant biloma with right heart compression in a patient undergoing hemodialysis

Abstract

A biloma is a localized bile collection caused by bile leakage that is usually resolved with percutaneous drainage. In most cases, clinical manifestations are limited to local symptoms such as abdominal pain and distension. Although bilomas can occasionally cause septic shock attributable to secondary infection or obstructive jaundice attributable to direct compression of the bile duct, cases involving heart compression by a biloma that leads to circulatory failure have been rarely reported. We report the case of a 79-year-old man undergoing maintenance hemodialysis who developed gallstone-associated cholecystitis complicated by gallbladder perforation that resulted in giant biloma formation that compressed the right heart and caused obstructive shock. At the onset of shock, the patient lacked fever or positive blood culture results, which are typical features of sepsis. The diagnosis of obstructive shock caused by direct compression of the right heart by a giant biloma was determined based on the findings of transthoracic echocardiography (collapsed right heart chambers) and computed tomography (massive fluid collection in the upper abdomen). After removing a large volume of bile using percutaneous transhepatic drainage, blood pressure and cardiac output rapidly improved. When patients undergoing hemodialysis present with unexplained shock and gallstone disease, clinicians should consider extracardiac compression by a giant biloma, promptly evaluate the cardiac and abdominal structures using echocardiography and computed tomography, respectively, and perform early drainage if necessary.

Introduction

A biloma is defined as a localized bile collection that forms outside the biliary tract as a result of abnormal bile leakage. The main causes include gallstone disease, iatrogenic injury associated with biliary surgery or percutaneous biliary drainage, and abdominal trauma [1]. Bilomas are relatively rare, with a reported incidence of 0.3–2.0% [2]. Percutaneous drainage is considered the first-line treatment of bilomas; favorable clinical outcomes are achieved in most cases [3]. Clinical manifestations of bilomas are nonspecific. In many patients, symptoms are limited to mild abdominal pain such as right upper quadrant pain or abdominal distension, wherein the clinical course varies [4]. Consequently, the diagnosis may be delayed in the absence of severe abdominal pain or overt peritoneal signs. Reports of bilomas that enlarge to the extent that they cause hemodynamic collapse by directly compressing the surrounding organs, particularly the heart or great vessels, are rare.

Obstructive shock is characterized by reduced cardiac output caused by mechanical impairment in venous return to the heart or obstruction of cardiac outflow. Typical causes include cardiac tamponade, pulmonary embolism, and tension pneumothorax. Compression of the right heart by an intra-abdominal bile collection is not commonly considered in the differential diagnosis.

We report the case of an older man undergoing maintenance hemodialysis who developed gallbladder perforation with gallstone-associated cholecystitis that resulted in a giant biloma formation that directly compressed the right heart and caused obstructive shock.

Case report

A 79-year-old man who had been undergoing maintenance hemodialysis for 3 years due to chronic kidney disease of unknown etiology presented to the emergency department of our hospital with a history of fever over several days. He was using cilostazol for peripheral artery disease in the lower extremities and had undergone implantation of a leadless pacemaker (VVI mode; pacing rate, 40 beats/min) 6 months previously owing to complete atrioventricular block.

Laboratory test results revealed elevated inflammatory markers, including a C-reactive protein level of 13.3 mg/dL and white blood cell of 12,300/μL. Noncontrast computed tomography (CT) revealed an impacted 7.2-mm gallstone at the gallbladder neck and gallbladder distension, which led to the diagnosis of acute calculous cholecystitis (Fig. 1a, b). Additionally, hypokalemia (3.3 mEq/L) and hypomagnesemia (1.6 mg/dL) with marked QTc prolongation to 618 ms were observed.

Fig. 1.

a, b Noncontrast abdominal computed tomography (CT) images on admission showing an impacted gallstone at the gallbladder neck with gallbladder distension. c Follow-up noncontrast CT image on day 46 demonstrating improvement of cholecystitis

After admission, polymorphic ventricular tachycardia consistent with torsades de pointes suddenly developed. The return of spontaneous circulation was achieved after 3 min of cardiopulmonary resuscitation. Subsequently, percutaneous transhepatic gallbladder aspiration was performed and yielded turbid bile. Correction of electrolyte abnormalities and meropenem were initiated. Blood and bile cultures at admission revealed extended-spectrum β-lactamase-producing Escherichia coli.

After confirming inflammatory marker improvement, the patient was weaned from mechanical ventilation on day 13. Follow-up CT scans on days 8 and 18 showed improvement in cholecystitis; meropenem was discontinued on day 25. The patient reported abdominal discomfort after recovery of consciousness; however, follow-up CT performed on day 46 showed improvement in cholecystitis. Additional antibiotic therapy was not administered and cholecystectomy was not performed (Fig. 1c). After day 46, transient episodes of hypotension were occasionally observed during hemodialysis; however, based on findings such as chest radiography, the patient’s dry weight was considered to be generally appropriate.

On day 53, sudden right upper quadrant pain developed without an apparent trigger and manifested with shock (blood pressure, 62/39 mmHg; heart rate, 66 beats/minute). Norepinephrine infusion was initiated; moreover, fever and an altered level of consciousness were not observed. Oxygen saturation was maintained at 96% on room air; the patient’s respiratory rate was 20 breaths/minute. Laboratory test results revealed an elevated C-reactive protein level (4.72 mg/dL) without elevated hepatobiliary enzymes (Table 1). Based on the pain location and clinical course, septic shock secondary to recurrent acute cholecystitis was suspected. Therefore, meropenem was restarted on the same day. Since the event occurred out of hours and immediate imaging resources were limited and the patient’s vital signs transiently stabilized after fluid resuscitation and vasopressor support, we planned to perform ultrasonography and CT on the following day.

Table 1.

Laboratory findings on day 53

Complete blood count
WBCs	3700/μL
Neutrophils	68.8%
Lymphocytes	12.3%
Monocytes	5.2%
Eosinophils	13.4%
Basophils	0.3%
Hb	8.4 g/dL
Ht	25.6%
Platelets	47,000/μL
Blood biochemical analysis
Total protein	4.6 g/dL
Albumin	1.8 g/dL
AST	24 U/L
ALT	5 U/L
LDH	240 U/L
Total bilirubin	1.1 mg/dL
Cholinesterase	39 U/L
CRP	4.7 mg/dL
Na	133 mEq/L
Cl	105 mEq/L
K	4.4 mEq/L
Blood urea nitrogen	19.8 mg/dL
Creatinine	3.73 mg/dL
Venous blood gas analysis (room air)
pH	7.386
PaCO2	32.8 mmHg
PaO2	34.2 mmHg
HCO3	19.3 mmol/L
Base excess	− 5.2 mmol/L
Lactate	1.20 mmol/L
Anion gap	8.7 mmol/L

ALT Alanine aminotransferase, AST Aspartate aminotransferase, Cl Chloride, CRP C-reactive protein, Hb Hemoglobin, Ht Hematocrit, K Potassium, LDH Lactate dehydrogenase, Na Sodium, WBCs White blood cells

PaCO₂ Carbon dioxide partial pressure arterial PaO₂ Oxygen partial pressure arterial

Since the typical features of septic shock, such as fever and an altered mental status, were absent, transthoracic echocardiography was performed the following day to investigate the cause of shock. A hypoechoic area was observed inferior to the heart; furthermore, the right heart chambers were collapsed due to direct external compression with reduced cardiac output (1.85 L/min) (Fig. 2a, b). Cardiac output was calculated by the velocity–time integral of the left ventricular outflow tract. Significant valvular disease and pericardial effusion were not observed.

Fig. 2.

a, b Transthoracic echocardiography images on day 54 showing a hypoechoic area surrounding the inferior and posterior aspects of the heart with collapse of the right heart chambers caused by direct external compression. c, d Contrast-enhanced computed tomography (CT) images on day 54 demonstrating a giant encapsulated 200- × 75- × 108-mm fluid collection adjacent to the liver consistent with a biloma. The location of the gallbladder stone remained unchanged. e, f Follow-up transthoracic echocardiography and noncontrast CT images after percutaneous drainage showing resolution of the fluid collection compressing the right heart and improvement in right heart chamber collapse. Ao, aorta; LA, left atrium; LV, left ventricle; RA, right atrium; RV right ventricle

Contrast-enhanced CT revealed a 200- × 75- × 108-mm fluid collection adjacent to the liver with a well-defined capsule but without internal contrast enhancement (Fig. 2d, e). Pulmonary embolism and aortic dissection were ruled out. A localized bile-containing fluid collection secondary to gallbladder perforation was suspected; therefore, percutaneous transhepatic drainage was performed. An 18-gauge needle was used for transhepatic puncture. A 6-Fr pigtail catheter placed in the lesion yielded approximately 1000 mL of pale yellow bile-like fluid (Fig. 3). Thereafter, blood pressure promptly increased. Since the patient was hemodynamically unstable due to shock, we judged that prompt drainage to relieve the causative compression should be prioritized over identifying the exact leak site. Therefore, we did not perform magnetic resonance cholangiopancreatography or endoscopic retrograde cholangiopancreatography to evaluate potential communication between the biloma and biliary tract. We also did not inject contrast through the drainage catheter, considering the potential risk of enlarging the fistulous tract due to increased pressure during contrast administration. Norepinephrine was discontinued the next day. Transthoracic echocardiography performed the day after drainage demonstrated resolution of the collapsed right heart chambers and improvement in cardiac output to 5.37 L/min (Fig. 2c). Blood cultures obtained prior to antibiotic initiation on day 53 and biloma fluid cultures collected at the time of drainage on day 54 revealed negative results. Drainage output was 48 mL on day 55; however, it ceased after day 56.

Fig. 3.

Bile-like fluid obtained by percutaneous transhepatic drainage. The fluid is pale yellow and shows no apparent signs of infection

Although the catheter was accidentally dislodged on day 58, noncontrast CT showed marked reduction in the bile collection; therefore, catheter reinsertion was not performed (Fig. 2f). Thereafter, bile collection did not recur. However, on day 63, methicillin-resistant coagulase-negative Staphylococcus bacteremia associated with a central venous catheter infection developed. Therefore, vancomycin was initiated and the catheter was removed. Nevertheless, continuation of hemodialysis became difficult due to malnutrition and chronic inflammation. The patient died on day 84.The clinical course is illustrated in Fig. 4.

Fig. 4.

Clinical course of the present case. Mean arterial pressure (MAP) throughout the course. CO, cardiac output; VT, ventricular tachycardia; ROSC, return of spontaneous circulation; PTGBA, percutaneous transhepatic gallbladder aspiration; TTE, transthoracic echocardiography; MR-CoNS, methicillin-resistant coagulase-negative Staphylococcus; RA, right atrium; RV right ventricle

Discussion

In the present case, gallstone disease and high-risk host-related factors were considered the main contributors to biloma development. The pathogenesis of a biloma generally involves bile leakage from a disrupted biliary tract that is followed by encapsulation by the surrounding tissues in response to peritoneal irritation, resulting in the formation of a localized bile collection. Since bile is highly chemically irritative, even a small amount of leakage can induce local inflammation and promote fibrous capsule formation [5].

In our patient, gallstone impaction at the gallbladder neck led to increased intraluminal pressure and subsequent development of acute cholecystitis. Gallbladder perforation during the clinical course was considered the direct cause of bile leakage. In this case, percutaneous transhepatic gallbladder aspiration was performed early after admission; therefore, an iatrogenic biloma needed to be considered in the differential diagnosis. In general, procedure-related bilomas most commonly develop within approximately 1–2 weeks after an intervention. However, in our patient, repeated non-contrast CT scans on days 8, 18, and 46 did not demonstrate any biloma. Moreover, the patient did not develop fever or worsening abdominal symptoms during the early post-procedural period; therefore, we considered an iatrogenic etiology less likely [6]. The leaked bile was likely rapidly encapsulated within the abdominal cavity and gradually enlarged as a localized bile collection adjacent to the liver after gallbladder perforation.

Gallstone disease is more prevalent (22–30%) in patients undergoing hemodialysis or peritoneal dialysis than in the general population [7, 8]. Limited studies have directly demonstrated thinning or fragility of the gallbladder wall in patients undergoing dialysis. However, several factors that increase the risk of gallbladder perforation, including advanced age, male sex, history of cholecystitis, and coexisting cardiovascular disease, were present in this case [9]. In addition, in patients undergoing hemodialysis, splanchnic hypoperfusion associated with ultrafiltration and intradialytic hypotension and a uremia-related bleeding tendency (i.e., platelet dysfunction) have been suggested to predispose to ischemic injury and necrosis of the gallbladder wall, potentially facilitating progression to perforation and biloma formation [10, 11]. In our patient, uremic neuropathy may attenuate pain perception, which could have delayed symptom recognition [12].

The patient initially presented with acute calculous cholecystitis. In Japan, acute cholecystitis management is guided by the Tokyo Guidelines 2018, which recommend treatment strategies based on the disease severity, surgical risk, and response to initial therapy. In this case, the patient experienced cardiac arrest caused by torsades de pointes at admission that was classified as grade III (severe) acute cholecystitis with associated circulatory dysfunction. In accordance with to the Tokyo Guidelines 2018, percutaneous transhepatic gallbladder aspiration was initially performed to enable source control; furthermore, elective cholecystectomy was considered [13]. However, the Charlson Comorbidity Index score was 6, which indicated a high perioperative risk; therefore, the treatment strategy focused on careful systemic management and reassessment of surgical indications [14]. Subsequent development of gallbladder perforation and giant biloma formation are important to determining the treatment strategy. In patients at high risk, continuous multidisciplinary evaluations that involve surgeons, anesthesiologists, and other specialists are essential for determining the optimal indication for and timing of cholecystectomy.

In the present case, the initial manifestations of the biloma were shock and right upper quadrant pain. Shock is generally classified as hypovolemic, cardiogenic, obstructive, or distributive [15]. Common causes of obstructive shock include cardiac tamponade, pulmonary embolism, and pneumothorax; however, obstructive shock caused by direct compression of the right heart by an intra-abdominal lesion is extremely rare [16]. Findings suggestive of hemorrhage or dehydration were not observed in our patient. Transthoracic echocardiography demonstrated preserved left ventricular systolic function; contrast-enhanced CT ruled out pulmonary embolism and aortic dissection. The negative results of the blood and bile cultures suggested the absence of infection within the bile collection. Inflammatory marker elevation can be considered the result of local inflammation associated with bile leakage [17]. Based on these findings, obstructive shock caused by direct compression of the right heart by a giant biloma was diagnosed.

A PubMed literature search (up to October 2025) using “biloma,” “obstructive shock,” “cardiac compression,” and “tamponade” as keywords did not yield previous reports of biloma-related extracardiac compression or shock. In contrast, several cases of large intra-abdominal cystic lesions, such as giant hepatic cysts, that directly compressed the heart and caused right heart collapse or heart failure have been reported [18, 19]. The present case likely represents a similar rare pathophysiological mechanism. The rapid improvement in blood pressure after percutaneous drainage and marked recovery of cardiac output observed with follow-up echocardiography strongly support this mechanism. However, the subsequent clinical course of this case was complicated by central venous catheter-related infection and was not attributable to the biloma itself.

In patients undergoing hemodialysis, acute circulatory failure can result from various causes, including infection, dehydration, heart failure, and arrhythmia. When shock develops in patients with underlying gallstone disease, septic shock is often the primary consideration. However, atypical clinical presentations, such as those comprising the absence of fever, preserved mental status, or negative blood culture results, structural causes should be actively investigated using transthoracic echocardiography and CT. In the present case, echocardiography was performed to evaluate the cause of shock and revealed right heart collapse, which subsequently led to the identification of a giant biloma on CT and prompt percutaneous drainage. This sequential diagnostic process highlights the importance of imaging when evaluating atypical shock.

In conclusion, when patients undergoing hemodialysis present with acute circulatory failure and gallstone disease, obstructive shock attributable to extracardiac compression caused by a giant biloma should be included in the differential diagnosis. Early assessments including echocardiography of the heart and abdominal CT followed by prompt decompressive drainage, when indicated, may comprise a life-saving intervention.

Authors' contribution

Hidemasa Oda contributed to the conception and design of the study and drafted the manuscript. Hidemasa Oda, Tomoki Ohashi, Atsuko Hayakawa, Hiroya Tanaka, Mikio Takagi, Shotaro Higuchi, and Hiroaki Kumazawa contributed to data acquisition, analysis, and interpretation. All authors critically revised the manuscript for important intellectual content. All authors approved the final version of the manuscript and agree to be accountable for all aspects of the work, ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Funding

Not applicable.

Data availability

Data sharing not applicable to this article as no datasets were generated or analysed during the current study.

Declarations

Competing interests

The authors have no relevant financial or non-financial interests to disclose.

Ethics approval

Ethics approval was not required as this is a case report. However, the study was performed in accordance with the ethical standards as laid down in the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards.

Consent to participate

Written informed consent was obtained from the patient.

Consent for publication

Written informed consent was obtained from the patient for the publication of his data.