Surg Today (2004) 34:973–976DOI 10.1007/s00595-004-2840-3

Acute Cholecystitis with a Hemocholecyst as an Unusual Presentationof Gallbladder Cancer: Report of a Case

Joseph Ku, Jacob DeLaRosa, Justin Kang, David Hoyt, and Raul Coimbra

Division of Trauma, Department of Surgery, University of California San Diego, School of Medicine, 200 West Arbor Drive, San Diego,CA 92103-8896, USA

Case Report

A 60-year-old moderately obese woman of Europeandescent presented to the emergency room with a 3-dayhistory of right upper quadrant pain. She denied havinghad any fevers or chills, but complained of nausea,vomiting, and anorexia. Before any radiographic study,her history appeared to correlate well with her physicalfindings, including right upper quadrant pain and a posi-tive Murphy’s sign. A routine emergency room ultra-sound of the right upper quadrant was done to confirmthe suspected diagnosis of acute cholecystitis (Fig. 1).The ultrasound showed multilobulated echogenicmaterial, along with the more classic signs of acutecholecystitis including a thickened gallbladder wall andpericholecystic fluid. No gallstones were seen, but a2.2-cm portocaval node was noted. The radiologist’sreport included sludge, intraluminal hemorrhage, anda fungating mass in the gallbladder wall.

A computed tomography (CT) scan was done toassist with the diagnosis, revealing an enlarged gall-bladder with apparent sludge and multiple enlargedperipancreatic, portal, and periaortic nodes (Fig. 2). Ahepatic cyst was incidentally discovered. A completeblood count and biochemical analysis failed to show anyabnormalities and the liver function tests showed onlya slightly elevated alkaline phosphatase level. Based onthe ultrasound findings of acute cholecystitis explainingthe patient’s symptoms, and the CT evidence of possiblemetastatic gallbladder cancer, open cholecystectomywas indicated without the need for any additional imag-ing studies.

Laparotomy revealed a large and distended gall-bladder (Fig. 3) from which a large amount of clottedblood was aspirated. We did not send the aspirate topathology because the result would not have changedthe intraoperative or postoperative treatment strate-gies. After an uncomplicated cholecystectomy, thegallbladder was opened and found to contain a large

AbstractSeveral atypical presentations of gallbladder carcinomahave been reported, but one of the rarest is intraluminalhemorrhage. We report a case of carcinoma of the gall-bladder disclosed by an emergency cholecystectomy,performed for acute cholecystitis caused by ahemocholecyst. The diagnostic approaches and charac-teristics of a hemocholecyst associated with carcinomaof the gallbladder are discussed.

Key words Hemocholecyst · Gallbladder carcinoma ·Cancer · Acute cholecystitis

Introduction

Gallbladder cancer is still associated with a poor prog-nosis.1 It is the most common malignancy of the biliarytract and the fifth most common malignancy of thedigestive system, with a reported incidence of 2.5 per100000 residents in the United States. The most com-mon symptoms of this disease are pain (76%), jaundice(38%), anorexia (32%), and weight loss (39%), butatypical presentations, such as empyema, acute chole-cystitis, post-cholecystectomy benign biliary stricture,gastric outlet obstruction, and liver abscess, have beenreported.2 Carcinoma of the gallbladder manifestingas a gallbladder intraluminal hemorrhage occurs inonly about 1% of patients. We report an unusual caseof carcinoma of the gallbladder diagnosed during anemergency cholecystectomy, performed for acutecholecystitis caused by a hemocholecyst. The diagnosticapproaches and characteristics of a hemocholecystassociated with carcinoma of the gallbladder arediscussed.

Reprint requests to: R. CoimbraReceived: May 12, 2003 / Accepted: March 9, 2004

974 J. Ku et al.: Hemocholecyst Associated with Gallbladder Cancer

fungating mass (Fig. 4). Grossly, the gallbladderweighed 102 g, measured 12.5 � 7.0 � 3.0cm, andcontained an exophytic necrotic lesion. Microscopic ex-amination revealed a moderately differentiated adeno-carcinoma (Fig. 5), with negative extension beyond theserosa. A celiac lymph node biopsy was also performed,which was positive for malignancy. Because of the ex-tent of the disease process and the evidence of lymphnode involvement, we did not perform a more radicaloperation to include wedge resection of the gallbladderbed and aggressive lymph node excision. The tumor wasstaged as T2N2MX. The patient had an uneventful post-operative course and was discharged 5 days after theoperation to undergo postoperative chemotherapy. Shewas seen 1 year postoperatively, but was then lost tofollow-up.

Fig. 1. Abdominal ultrasound showed multilobulated echo-genic material in the gallbladder, a thickened gallbladder wall,and pericholecystic fluid. No gallstones were seen

Fig. 2. Abdominal computed tomography showed an en-larged gallbladder with sludge. A hepatic cyst was notedincidentally

Fig. 3. Intraoperative photograph showing a large and dis-tended gallbladder

Fig. 4. Intraoperative photograph showing the opened gall-bladder. A large fungating mass was found in the body of thegallbladder

Fig. 5. Histopathological examination revealed moderatelydifferentiated adenocarcinoma of the gallbladder (�400)

975J. Ku et al.: Hemocholecyst Associated with Gallbladder Cancer

Discussion

Gallbladder cancer has an incidence of 2.5 per 100000in the United States,1 the lowest incidence being seenamong African Americans and a higher incidenceamong Mexican and Native Americans.2 This diseasemainly affects women over the age of 65 years. Environ-mental risk factors have not been reported, but itsassociation with cholelithiasis and porcelain gallbladderis well established.2 After elective operations for gall-stones, tumors are discovered incidentally in less than1% of patients, although it is known that stones largerthan 3cm in diameter carry a 10-fold increase in the riskfor cancer.3 Most series have reported a 5-year survivalrate of less than 5%. Current thinking suggests gall-stones and carcinoma probably occur secondary to achronic inflammatory state. Gallbladder cancer was firstdescribed in 1877 by Macillian de Stoll, despite which itis still extremely difficult to diagnose early. Even withadvances in imaging modalities to aid in the detectionof occult malignancies, the options for diagnosing andtreating gallbladder cancer fall short of what one wouldexpect. Macillian’s initial description still stands true:“. . . the prognosis of patients with this disease is dismal.”Even today, the preoperative diagnosis of gallbladdercancer is rare, and in most cases it is found intraopera-tively or at autopsy.

Many authors have described different presentationsof gallbladder cancer, including empyema, cholecystitis,benign biliary stricture, liver abscess, gastric outletobstruction, and carcinoma of the head of the pancreas.A hemocholecyst is a very unusual presentation ofgallbladder cancer, and is therefore not well docu-mented. In 1892, Naunyn described three cases of ahemocholecyst caused by ruptured aneurysms of eitherthe hepatic or cystic artery in relation to gallstones orcholecystitis.4 Hemocholecyst has since been defined ashemorrhage into the gallbladder, which does not resultin rupture of the gallbladder wall. This entity is uniqueand not necessarily associated with hemobilia. Sinceits first description, there have been several reports ofhemocholecyst, most of which were related to infectiousor chronic inflammatory disorders of the gallbladder.5

There have also been reports of acute cholecystitis be-ing caused by hemorrhage secondary to traumatic acci-dents.6 A recent review of the literature by Heise et al.7

examined the different causes of blood accumulatingin the gallbladder. In most cases, the bleeding appearedto come from the liver, and was caused by trauma. Withthe advent of more invasive, nonoperative procedures,iatrogenic trauma has also come to be a concern.8 How-ever, direct causes of hemocholecyst are more limited,and include malignancy, cholelithiasis, vascular disor-ders, and the presence of heterotopic gastrointestinalmucosa in the gallbladder. Malignant sources include

primary tumors, metastasis from a distant site, or inva-sion from an adjacent liver tumor. The possible mecha-nisms for the development of hemocholecysts includedirect invasion of a tumor to either the liver or avessel in its proximity, or it can be caused by afungating necrotic mass with local extension, as in ourpatient.

The term “hemocholecyst” is still often misused andone must recognize that hemobilia and hemocholecystare two distinct entities. In discussing a case of gallblad-der cancer in a patient without signs or symptoms ofupper gastrointestinal bleeding, Uchiyama et al.9 exam-ined the causes of hemobilia, including trauma (39%),surgery (17%), calculi (15%), inflammation (13%),vascular disorders (11%), and neoplasms (5%). Theystated correctly that hemobilia with gallbladder canceris rare; however, our interpretation of that case reportis that the patient probably had a hemocholecyst ratherthan hemobilia.

Gallbladder cancer presenting as acute cholecystitisassociated with a hemocholecyst is rare and poorlydescribed in the literature. Gimmon et al.10 reporteda similar case, although acute cholecystitis occurredsecondary to obstruction of the cystic duct by the tumoritself. Conversely, our patient suffered symptoms ofacute cholecystitis secondary to obstruction of the cysticduct by blood clots, although the tumor was distantfrom it. This presentation is especially unusual, consid-ering the large amount of fibrinogen present in the bile,which is thought to prevent complete obstruction of thecystic duct by clotted blood.11

Gallbladder cancer is classically diagnosed by theso-called porcelain gallbladder, created by calcification,which is easily recognized on an abdominal plain film.Currently, the best imaging modalities for diagnosingthese tumors are ultrasound and CT scans. Ultrasoundhas been used for a long time in the diagnosis of acutecholecystitis. Unfortunately, polyps and carcinomashave echogenicity similar to the gallbladder wall, mak-ing it difficult to distinguish them from a thickened wallsecondary to acute inflammatory changes. Endoscopicultrasound can assist in distinguishing benign frommalignant disease, but given its more invasive nature, itis not used routinely. On the other hand, CT carrieshigh sensitivity and specificity in the identification of agallbladder mass, but lower sensitivity in identifyingnodal spread, and is therefore not considered ideal forstaging.6 Angiography has also been used in some cen-ters with questionable efficacy. More recently, magneticresonance cholangiopancreatography has evolved intoa very sensitive and specific technique that can also helpin the staging process. However, in the presence of ahemocholecyst all these imaging modalities become lesssensitive. Given the rarity of gallbladder cancer, and theeven rarer association with a hemocholecyst, prompt

976 J. Ku et al.: Hemocholecyst Associated with Gallbladder Cancer

diagnosis still eludes the average radiologist, surgeon,and clinician.

Complete resection of the tumor provides the onlychance of cure, but an early preoperative diagnosis isessential. In fact, the 5-year survival rate after resectionwith curative intent has been reported at less than 17%.3

The extent of operative intervention for different stagesof gallbladder cancer remains controversial. The cur-rent recommendations are based on the primary tumoras defined by the American Joint Committee on Can-cer. Shirai and Yoshida reported that simple cholecys-tectomy was curative in most patients with T1 cancers,and 40% survival was even achieved in patients withpT2 cancer.12 Therefore, extended survival has beenachieved in patients with higher tumor stages. A moreradical second operation, which often includes wedgeresection of the gallbladder bed and resection of thesupraduodenal segment of the bile duct, with en blocdissection of the regional lymph nodes, has been shownto increase 5-year survival in patients with pT2 cancer toup to 90%. On the other hand, no such survival benefitwas seen after reoperations in patients with pT3 or pT4tumors.

Adjuvant chemotherapy has not proven effectiveagainst gallbladder carcinoma, although most studieswere limited by the small number of patients enrolled.The most extensively studied chemotherapeutic agentis 5-fluorouracil, associated with a response rate of only10%–24%.13

In conclusion, we described a hemocholecyst causingacute cholecystitis as a very rare presentation of gall-bladder cancer. To our knowledge, only one other suchcase has ever been described.4 Until we can consistentlydiagnose gallbladder cancer preoperatively at an earlystage, it will continue to carry high 5-year mortalityrates. To diagnose this devastating disease earlier in itscourse, radiologists and clinicians need to be aware ofall of its different presentations. As improvements are

made in diagnostic modalities and we become moreaware of the possibility of this disease, we will be closerto achieving a more acceptable outcome for thesepatients.

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