Case Report Open Access
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 26, 2022; 10(27): 9734-9742
Published online Sep 26, 2022. doi: 10.12998/wjcc.v10.i27.9734
Gallbladder hemorrhage–An uncommon surgical emergency: A case report
Maria Rosaria Valenti, Andrea Cavallaro, Maria Di Vita, Antonio Zanghi, Giovanni Longo Trischitta, Alessandro Cappellani
Maria Rosaria Valenti, Andrea Cavallaro, Maria Di Vita, Antonio Zanghi, Giovanni Longo Trischitta, Alessandro Cappellani, Department of Surgery, University of Catania Medical School, University of Catania, Catania 95123, Italy
ORCID number: Maria Rosaria Valenti (0000-0003-4185-1816); Andrea Cavallaro (0000-0002-0311-5191); Maria Di Vita (0000-0002-5578-2834); Antonio Zanghi (0000-0002-7765-9626); Giovanni Longo Trischitta (0000-0001-5958-5638); Alessandro Cappellani (0000-0002-9113-2396).
Author contributions: All authors discussed the results and contributed to the final manuscript.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: All authors report no relevant conflict of interest for this article.
CARE Checklist (2016) statement: The authors have read the Care Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Andrea Cavallaro, MD, PhD, Associate Research Scientist, Doctor, Medical Assistant, Surgeon, Surgical Oncologist, Teaching Assistant, Department of Surgery, University of Catania Medical School, University of Catania, Via S. Sofia 78, Catania 95123, Italy. andreacavallaro@tiscali.it
Received: May 10, 2021
Peer-review started: May 10, 2021
First decision: June 5, 2021
Revised: June 14, 2021
Accepted: June 30, 2022
Article in press: June 30, 2022
Published online: September 26, 2022

Abstract
BACKGROUND

Gallbladder hemorrhage is a life-threatening disorder. Trauma (accidental or iatrogenic such as a percutaneous biopsy or cholecystectomy surgery), cholelithiasis, biliary tract parasitosis, vasculitis, vascular malformations, autoimmune and neoplastic diseases and coagulopathies have been described as causes of hemorrhage within the lumen of the gallbladder. The use of non-steroidal anti-inflammatory drugs and anticoagulants may represent a risk factor.

CASE SUMMARY

We report the case of a 76-year-old male patient. An urgent contrast computed tomography scan demonstrated relevant distension of the gallbladder filled with hyperdense non-homogeneous content. The gallbladder walls were of regular thickness. Near the anterior wall a focus of suspected active bleeding was observed. Due to the progressive decrease in hemoglobin despite three blood transfusions, this was an indication for urgent surgery.

CONCLUSION

Early diagnosis of this potentially fatal pathology is essential in order to plan a strategy and eventually proceed with urgent surgical treatment.

Key Words: Gallbladder, Hemorrhage, Anticoagulants, Cholecystectomy, Surgery, Case report

Core Tip: Gallbladder hemorrhage is an uncommon life-threatening disorder. There are many causes of this condition: trauma, cholelithiasis, biliary tract parasitosis, vasculitis, vascular malformations, autoimmune and neoplastic diseases and coagulopathies. We report the case of a 76-year-old male patient. An urgent contrast computed tomography scan demonstrated relevant distension of the gallbladder filled with hyperdense non-homogeneous content and a focus of suspected active bleeding. The patient underwent urgent surgery to stop the hemorrhage. Early diagnosis of this insidious and potentially fatal pathology is essential to plan the best treatment strategy for patients.



INTRODUCTION

Gallbladder hemorrhage is a rare condition, which can be difficult to diagnose. It manifests symptoms present in other more common pathologies, such as fever, nausea, abdominal pain, and Murphy's sign. Trauma (accidental or iatrogenic such as a percutaneous biopsy or cholecystectomy surgery), cholelithiasis, biliary tract parasitosis (e.g., ascariasis), vasculitis, vascular malformations, neoplastic diseases, and coagulopathies have been described as causes of hemorrhage within the lumen of the gallbladder. The use of non-steroidal anti-inflammatory drugs (NSAIDs) and anticoagulants may represent a risk factor. During the evolution of flogosis, necrosis of the gallbladder mucosa may result in bleeding from the vessels located within the organ wall. The use of imaging methods such as ultrasound, computed tomography (CT) and angiography can be useful in diagnosing this uncommon condition. As a medical emergency with a reported mortality rate of 15%-20%, early diagnosis is mandatory and can result in a better outcome for the patient[1-4]. We describe the case of a patient treated with NSAIDs and anticoagulants, who developed severe anemia due to intracolecystic hemorrhage that required urgent surgery.

CASE PRESENTATION
Chief complaints

The patient complained of abdominal pain and constipation.

History of present illness

We report the case of a 76-year-old male patient who attended the emergency room due to abdominal pain and constipation.

History of past illness

In the anamnesis he presented osteoporosis, parkinsonism, vertebral stabilization (metal plates and screws) for L2-L3 arthrodesis (2016), L1-L2 spondylodiscitis, chronic pain in the lumbar region, stiffness in the upper limbs and lower limbs, small steps and impairment of the extensor muscles of the spinal column with bent spine syndrome. In July 2020, due to trauma, he attended the emergency room, where, lacking diagnostic evidence, he was discharged and transferred to a rehabilitation institution. He had been taking the following drugs for the last few years at home: Pantoprazole, CardioASA, Bromazepam, Durogesic, and Cardicor.

Personal and family history

No relevant personal and family history.

Physical examination

On physical examination, the patient appeared oriented, cooperative, eupnoic, malnourished, with muscle atrophy and widespread hypotonia. Abdominal examination demonstrated diffuse abdominal pain. His heart rate was 68 bpm, blood pressure was 120/70 mmHg, and temperature was 36°C.

Laboratory examinations

Blood chemistry showed the following: hemoglobin 11.2 g/dL; white blood cell count 10.790/μL; total bilirubin 0.50 mg/dL; aspartate aminotransferase 40 U/L; alanine aminotransferase 28 U/L; alkaline phosphatase 115 U/L; amylase 406 U/L; C-reactive protein: 150 mg/L.

Imaging examinations

To assess the suspicion of chronic pancreatitis, the patient underwent abdominal ultrasound, and the pancreas showed multiple calcifications in the parenchyma. Moreover, the aorta demonstrated an irregular caliber with progressive stenosis. This finding required an in-depth study; therefore, CT angiography was performed and the common right artery demonstrated CT signs of dissection and ulcerated atheromatous plaque at the origin. Dilated gallbladder, slightly dilated extrahepatic biliary tract, increased volume in the pancreatic gland with small hypodense formations in the pancreas head (maximum diameter 3 mm) were also highlighted. Due to the finding of ulcerated atheromatous plaque, Fondaparinux 2.5 mg/d was administered as suggested by the vascular surgeon consultant.

However, the persistence of continuous lumbar pain led the patient to NSAIDs and morphine-like analgesic self-administration, the dosage of which was increased and reduced according to the patient's symptoms relief. Approximately 10 d after Fondaparinux administration, the patient suddenly experienced severe anemia, hyperbilirubinemia, increased cholestasis and transaminase. On physical examination the abdomen was painless. No blood was found in the stool. An urgent abdominal ultrasound, with the patient still in bed, was performed. Evidence of distended gallbladder filled with non-homogeneous hyperechoic material and a slightly dilated intrahepatic biliary tract were observed (Figure 1). The common bile duct was not visible due to intestinal gas.

Figure 1
Figure 1 Ultrasound scan. Distended gallbladder filled with non-homogeneous hyperechoic material and slightly dilated intrahepatic biliary tract, the common bile duct was not visible due to intestinal gas.

Therefore, the patient underwent an urgent CT scan, which demonstrated relevant distension of the gallbladder filled with hyperdense non-homogeneous content. The gallbladder walls were of regular thickness. Near the anterior wall, a focus of suspected active bleeding was noted. Intra- and extra-hepatic biliary ducts demonstrated wider dilatation when compared to the previous CT scan (Figure 2).

Figure 2
Figure 2 Computed tomography scan of intra- and extra-hepatic biliary ducts demonstrated wider dilatation.
FINAL DIAGNOSIS

Gallbladder hemorrhage.

TREATMENT

Our hospital is an emergency referring center, with multidisciplinary expertise readily available. An interventional radiological consultation was sought with the aim of evaluating the risk-benefit ratio of cystic artery embolization and/or cholecystostomy. The risk of gallbladder necrosis due to cystic artery occlusion and the risk of hemoperitoneum due to percutaneous drainage led the surgical team to select upfront surgery. The decrease in hemoglobin despite three blood transfusions, coagulation disorders and worsening of his general condition required an effective and timely solution. Therefore, the patient underwent urgent surgery. Open cholecystectomy was performed. Choledocotomy with Kehr tube apposition completed the surgery due to the presence of dilated hepatocoledocus (approximately 25 mm) (Figure 3A). When the gallbladder was inspected at the backtable, it appeared entirely occupied by clots (Figure 3B).

Figure 3
Figure 3 Surgical specimen. A: When open cholecystectomy was performed, choledocotomy with Kehr tube apposition completed the surgery due to dilated hepatocoledocus (approximately 25 mm); B: When the gallbladder was inspected at the backtable, it appeared entirely occupied by clots.
OUTCOME AND FOLLOW-UP

A further blood transfusion, plasma and supportive medical therapy were administered during the perioperative period. The patient had a regular post-operative course until discharge. The T tube was removed 50 d after surgery. Histological examination demonstrated acute lithiasic cholecystitis without any relevant finding.

DISCUSSION

Gallbladder hemorrhage is a rare complication of cholelithiasis, and is difficult to diagnose due to the non-specificity of the symptoms, which may easily lead to possible thoracic aortic dissection for back pain or acute cholecystitis for right hypochondrium pain. It can also manifest with fever, nausea, jaundice, melena and increased indices of inflammation and markers of liver damage in blood tests (neutrophilic leukocytosis, hypertransaminasemia, hyperbilirubinemia)[1-4].

Among the causes of gallbladder hemorrhage, trauma, neoplasms of the biliary tract, lithiasic cholecystitis, parasitosis, vasculitis, autoimmune diseases, and primary or secondary coagulopathies (e.g., liver cirrhosis, renal failure) have been reported[5-10]. Most patients diagnosed with gallbladder hemorrhage have comorbidities and most take anticoagulants and NSAIDs. To date, approximately 51 case reports have been reported in the literature since 1980[5]. We performed a brief revision of the cases reported in the literature, and their treatment strategies (Table 1). Among the reports, over 80% of patients underwent surgery with cholecystectomy. Of these, 6/45 patients underwent elective laparoscopic cholecystectomy after conservative treatment. Open surgery was dominant in the urgent setting (24 vs 15 patients), and we could hypothesize that this surgical technique was chosen with the aim of better evaluation and control of extra-gallbladder sources of hemorrhage.

Table 1 Case reports in the literature since 1980.
Ref.
Journal
Patient age/gender
Anti-coagulation
Treatment choice
Nguyen D et al[16], 2021Journal of Radiology Case ReportsNCystic artery embolization, cholecystectomy
Chen X et al[17], 2021Hepatobiliary Surgery and Nutrition63 FNot mentionedERCP and ENBD, cholecystectomy
Leaning[18], 2021Journal of Surgical Case Reports73 MY-ApixabanLaparoscopic Cholecystectomy
Azam et al[19], 2021Journal of the National Medical Association55 MY-ApixabanCholecystectomy
Yam et al[2], 2020Radiology Case Reports51 FNCystic artery embolization, cholecystostomy, open cholecystectomy
Gomes et al[20], 2020BMJ Case Reports 87 MY-AspirinOpen cholecystectomy
Kishimoto et al[21], 2020Gan To Kagaku Ryoho. Cancer and Chemotherapy96 FNLaparoscopic cholecystectomy
Tarazi et al[5], 2019Journal of Surgical Case Reports87 MY-WarfarinCholecystostomy
65 FY-WarfarinConservative with IV antibiotics
92 FNCholecystostomy
Reens et al[22], 2019The Journal of Emergency Medicine76 MY-WarfarinCholecystostomy
Itagaki et al[23], 2019Journal of Medical Case Reports86 FY-EdoxabanConservative with IV antibiotics, elective laparoscopic cholecystectomy
Honda et al[24], 2019Journal of Clinical Rheumatology: practical reports on rheumatic & musculoskeletal diseases.71 MNLaparoscopic cholecystectomy
San Juan López C et al[25], 2019Revista espanola de enfermedades digestivas: organo oficial de la Sociedad Espanola de Patología Digestiva.55 MNLaparoscopic cholecystectomy
Ng et al[26], 2018BMJ Case Reports68 FNOpen cholecystectomy
Liefman et al[27], 2018International Annals of Medicine73 FY–RivaroxabanConservative with IV antibiotics, elective laparoscopic cholecystectomy
López et al[28], 2018Radiology84 MNot mentionedLaparoscopic cholecystectomy
Berndtson et al[29], 2017Surgical Infections Case Reports75 FNOpen cholecystectomy
Choi et al[30], 2017Trauma Image and Procedure65 MNLaparotomy + open cholecystectomy
Kinnear et al[31], 2017BMJ Case Reports74 MY- ApixabanLaparotomy + open cholecystectomy
Sishida et al[32], 2017Case Reports in Gastroenterology79 MY-Heparin for dialysisERCP and ENBD
Oshiro et al[33], 2017International Surgery61 FY – WarfarinConservative with IV antibiotics, elective laparoscopic cholecystectomy
Yoshida et al[34], 2017J-Stage73 MYLaparoscopic cholecystectomy
Tsai et al[35], 2016Medicine80 MNCholecystostomy, elective laparoscopic cholecystectomy
Calvo Espino et al[36], 2016Cirugía Española59 MNLaparotomy + Open cholecystectomy
Cho et al[37], 2015Korean Journal of Thoracic and Cardiovascular Surgery61 MY-WarfarinCholecystostomy drainage
Aljiffry et al[38], 2014Journal of Surgical Case Reports57 MNCystic artery embolization + open cholecystectomy
Onozawa et al[39], 2014International Surgery58 FNLaparoscopic cholecystectomy
Matsukiyo et al[40], 2014J-Stage68 FY-thrombolysisLaparotomy + open cholecystectomy
Seok et al[41], 2013Korean Journal of Internal Medicine84 MNLaparoscopic cholecystectomy
Taniguchi et al[42], 2013J-Stage48 MY-Heparin for dialysisLaparotomy + open cholecystectomy
Choi[43], 2012Zeitschrift für Gastroenterologie36 MY-Aspirin and ClopidogrelLaparoscopic cholecystectomy
Kwon et al[1], 2012Korean Journal of Hepatobiliary Pancreatic Surgery75 MY-WarfarinLaparoscopic cholecystectomy
Perez et al[10], 2011Revista Española De Enfermedades digestivas24 FNLaparoscopic to open cholecystectomy + intra-operative cholangiography
Jung et al[44], 2011Journal of the Korean Surgical Society55 MNLaparoscopic cholecystectomy
Parekh et al[7], 2010JAMA Surgery60 MNERCP + Laparoscopic cholecystectomy
50 MNLaparoscopic to open cholecystectomy
Lin et al[45], 2010Journal of Internal Medicine of Taiwan80 MY-WarfarinLaparoscopic cholecystectomy
Chen et al[46], 2010The American Journal of the Medical SciencesElderly MY-HeparinLaparoscopic cholecystectomy
Miyamoto et al[5], 2009J-Stage42 FNConservative with IV antibiotics, elective laparoscopic cholecystectomy
Oh et al[47], 2009Journal of the Korean Society of Magnetic Resonance in Medicine40 MNot mentionedLaparoscopic cholecystectomy
Lai et al[8], 2009Journal of Chinese Medical Association81 MY-Heparin for dialysisConservative with IV antibiotics, elective laparoscopic cholecystectomy
Morris et al[48], 2008Case Reports in Gastroenterology91 FNOpen cholecystectomy
Pandya et al[6], 2008Abdominal Imaging85 FY-WarfarinConservative with IV antibiotics
Kim et al[49], 2007World Journal of Gastroenterology55 MNCholecystostomy drainage
Gremmels et al[50], 2004Journal of Ultrasound in Medicine66 MNLaparotomy + open cholecystectomy
Hanaki et al[5], 2000J-Stage66 MNot mentionedLaparotomy + open cholecystectomy
Nishiwaki et al[51], 1999Journal of Gastroenterology58 MNLaparotomy + open cholecystectomy
Stempel et al[14], 1993Journal of Vascular and Interventional Radiology78 MY-Heparin during AAA repairCholecystostomy drainage
Brady et al[9], 1985Disease of the Colon & Rectum79 MNOpen cholecystectomy
Berland et al[52], 1980Journal of Computed Assisted Tomography56 MNLaparotomy + open cholecystectomy

The elevated prevalence (47%) of patients treated with antiplatelet agents and/or anticoagulants clearly underlines these drugs as risk factors. However, the role of other causes of hemorrhage (accidental or iatrogenic trauma, cholelithiasis, neoplasm, vascular anomalies and coagulopathies) in patients who did not take the aforementioned drugs is not insignificant.

Finally, we can assume that the incidence of this rare pathology is somehow underestimated, given the small number of cases in the literature. In the case described in this report, the patient had been taking cardioaspirin at home.

Moreover, the finding of dissection of the right iliac artery and ulcerated atheromatous plaque, and the thromboembolic risk derived from the patient's bed rest due to chronic lumbar pain suggested the administration of low molecular weight heparin. The self-administration and potential abuse of NSAIDs may have represented an additional risk factor.

Cholelithiasis and the intake of antithrombotic drugs may have played a primary role in the etiology of gallbladder hemorrhage. The damage caused by gallbladder mucosal stones usually heals spontaneously, but this may not occur in patients taking anticoagulants, creating blood oozing that can result in acute bleeding. The patient's medical history, physical examination, laboratory tests and radiological imaging are relevant to the diagnosis, to exclude other pathologies[11,12], in order to promptly plan a strategy, as gallbladder hemorrhage represents a potentially fatal surgical emergency. An initial evaluation with ultrasound can be carried out. Most cases of gallbladder hemorrhage demonstrate ultrasound features not common in acute cholecystitis.

The sonographic findings in hemorrhagic cholecystitis include focal wall thickening, intraluminal membranes and non-shadowing, non-mobile intraluminal echogenic material. There may be some echogenic layering material for which the differential diagnosis includes sludge[13]. The suspicion can be further confirmed by CT examination, which may demonstrate high attenuation within the gallbladder lumen with layering high attenuation fluid-fluid level representing blood or sludge. An early phase contrast-enhanced CT helps to detect active extravasation of contrast and blush within the lumen of the gallbladder[5,6].

The most suitable treatment for gallbladder bleeding is urgent laparoscopic or laparotomic cholecystectomy. In some selected cases, it is possible to plan a non-interventional strategy with antibiotic therapy and supportive medical therapy, postponing subsequent cholecystectomy surgery[5,10].

Rarely, in the case of patients ineligible for surgery, a percutaneous cholecystostomy may be indicated[14,15]. In our case, given the patient’s sudden anemia, despite blood transfusions and supportive medical therapy, due to the persistence of hemodynamic instability we proceeded with urgent surgery.

CONCLUSION

Gallbladder hemorrhage is a life-threatening complication of cholelithiasis. Early diagnosis of this potentially fatal pathology is essential in order to plan a treatment strategy and eventually proceed with urgent surgical treatment, to ensure timely life-saving decisions and the best results for the patient.

Footnotes

Provenance and peer review: Unsolicited article; Externally peer reviewed.

Peer-review model: Single blind

Specialty type: Surgery

Country/Territory of origin: Italy

Peer-review report’s scientific quality classification

Grade A (Excellent): 0

Grade B (Very good): 0

Grade C (Good): 0

Grade D (Fair): D, D

Grade E (Poor): 0

P-Reviewer: Kai K, Japan; Yasukawa K, Japan S-Editor: Wu YXJ L-Editor: Webster JR P-Editor: Wu YXJ

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