Xanthogranulomatous cholecystitis: report of a case and literature review on clinical differentiating factors from gallbladder carcinoma

Xanthogranulomatous cholecystitis (XGC) is a rare, benign, destructive inflammatory disease of the gallbladder that is assumed to be a variant of chronic cholecystitis. We herein present a rare case of XGC, which simulates gallbladder carcinoma with extensive involvement of the liver, omentum and the biliary trees. At surgery, total cholecystectomy with partial hepatectomy of the gallbladder bed and excision of adjacent xanthogranulomatous tissue was performed, but bilio‐enteric anastomosis for biliary decompression, which was the procedure preoperatively planned, was impossible to indicate because the common bile duct could not be clearly exposed by its infiltration showing mass formation. Therefore, retrograde transhepatic biliary drainage was eventually indicated for subsequent endoscopic therapy using stent placement to deal with the biliary structure caused by XGC. The patient has been leading a normal life after stent placement in the biliary tract for 6 months duration without any symptoms suggesting biliary stricture. In conclusion, XGC can simulate gallbladder cancer in its clinical presentation, radiological findings and even gross operative features. It is important to make preoperative ultrasound‐guided fine‐needle aspiration cytological diagnosis or intraoperative pathological diagnosis in order to avoid misdiagnosis and unnecessary therapy. Cholecystectomy, excision of adjacent xanthogranulomatous tissue, which often includes partial hepatic resection, are still the best management of XGC.     

Xanthogranulomatous cholecystitis expressing high levels of DUPAN-II

We report a case of xanthogranulomatous cholecystitis (XGC) showing high levels of serum DUPAN-II in a 65-year-old woman. Preoperative radiologic examination showed no abnormal findings except in the gallbladder. Endoscopic ultrasonography was effective for differentiating chronic cholecystitis from gallbladder cancer before the operation. Cholecystectomy was performed by laparotomy, and the diagnosis of XGC was confirmed intraoperatively by examining a frozen section. Histologically, no cancer lesion was observed in the gallbladder, while immunochemical reactivity to DUPAN-II was demonstrated in the brush-border area of the epithelium and in histiocytes in the gallbladder. The half-life of serum DUPAN-II in our patient after cholecystectomy was approximately 1 month, and finally dropped to within the normal range after cholecystectomy.     

La cholécystite xanthogranulomateuse, un diagnostic histologique inattendu: à propos d’un cas

Xanthogranulomatous cholecystitis (XC) is an uncommon form of chronic inflammation of the gallbladder characterized by a thickening of the gallbladder wall. It may clinically and radiographically mimic gallbladder carcinoma. In spite the progress of imagery, preoperative diagnosis is still often difficult which can lead to inappropriate extended surgery. We herein report the clinical presentation of a 71-year-old man with no medical history who was admitted to our department with a chief complaint of intermittent pain in his right upper abdomen, radiating to the back since a year. There was no history of jaundice, fever, anorexia, or weight loss. Physical examination revealed no signs of cholecystitis. Routine laboratory tests including white cell counts were within normal ranges. For further examination, we performed an abdominal ultrasonography. It revealed a thickening of the gallbladder wall on the hepatic side. There was no associated biliary dilatation or gallstones. CT scan of the abdomen showed similar findings with regional enlarged lymph nodes. The boundary of the gallbladder and liver parenchyma was ill defined. The patient was discharged in good condition on the 7^th day following surgery. He is now in good health eight months after surgery. As imaging findings were strongly suggestive of malignancy, we decided to perform a radical surgery. Peroperative finding shows a gallbladder mass invading the liver bed with some regional lymph nodes. We performed subsegmentectomy of the liver S4a + S5 and lymph node dissection. Histological examination showed diffuse chronic inflammatory cell infiltration with focal lipid-laden macrophage accumulation in the gallbladder wall and conclude on chronic xanthogranulomatous cholecystitis with no evidence of malignancy. The patient was discharged in good condition on the 7th day following surgery. He is now in good health 8 months after surgery. Xanthogranulomatous cholecystitis is a rare destructive inflammatory disease of the gallbladder which may be indistinguishable from a malignant neoplasm on the basis of imaging findings. The clinical presentation is similar to that of cholecystitis. A correct preoperative diagnosis of XC is important to avoid unnecessary laparotomy.     

Xanthogranulomatous cholecystitis: case series in a rural area, clinicopathological study and review of literature

Xanthogranulomatous cholecystitis is a destructive inflammatory disease of the gallbladder, rarely involving adjacent organs and mimicking an advanced gallbladder carcinoma. The diagnosis is usually possible only after pathological examination. We are reporting two of such rare cases in female patients attending our institute. In both patients xanthogranulomatous cholecystitis was diagnosed on histopathology.     

Xanthogranulomatous cholecystitis mimicking gallbladder carcinoma: An analysis of 42 cases

AIM: To review and evaluate the diagnostic dilemma of xanthogranulomatous cholecystitis (XGC) clinically.METHODS: From July 2008 to June 2014, a total of 142 cases of pathologically diagnosed XGC were reviewed at our hospital, among which 42 were misdiagnosed as gallbladder carcinoma (GBC) based on preoperative radiographs and/or intra-operative findings. The clinical characteristics, preoperative imaging, intra-operative findings, frozen section (FS) analysis and surgical procedure data of these patients were collected and analyzed.RESULTS: The most common clinical syndrome in these 42 patients was chronic cholecystitis, followed by acute cholecystitis. Seven (17%) cases presented with mild jaundice without choledocholithiasis. Thirty-five (83%) cases presented with heterogeneous enhancement within thickened gallbladder walls on imaging, and 29 (69%) cases presented with abnormal enhancement in hepatic parenchyma neighboring the gallbladder, which indicated hepatic infiltration. Intra-operatively, adhesions to adjacent organs were observed in 40 (95.2%) cases, including the duodenum, colon and stomach. Thirty cases underwent FS analysis and the remainder did not. The accuracy rate of FS was 93%, and that of surgeon’s macroscopic diagnosis was 50%. Six cases were misidentified as GBC by surgeon’s macroscopic examination and underwent aggressive surgical treatment. No statistical difference was encountered in the incidence of postoperative complications between total cholecystectomy and subtotal cholecystectomy groups (21% vs 20%, P > 0.05).CONCLUSION: Neither clinical manifestations and laboratory tests nor radiological methods provide a practical and effective standard in the differential diagnosis between XGC and GBC.     

Xanthogranulomatous Cholecystitis

Xanthogranulomatous cholecystitis exists in asmall but significant proportion of routinecholecystectomy specimens. A few recent reports haveshown a possible association of this disease withcarcinoma of the gallbladder. All cholecystectomizedspecimens were prospectively evaluated over a period oftwo and half years in a single surgical unit to examinethe incidence of xanthogranulomatous cholecystitis and its association, if any, with carcinoma ofthe gallbladder in an area that is prone to gallbladderdiseases. A total of 460 cholecystectomies wereperformed for various gallbladder diseases. Histological confirmation revealed chronic cholecystitis in311 (67.6%) cases, carcinoma of the gallbladder in 62(13.5%), acute cholecystitis in 29 (6.3%),xanthogranulomatous cholecystitis in 41 (8.9%), andxanthogranuloma and carcinoma of the gallbladder in one case(0.2%) only. Almost all cases were suspected to havechronic cholecystitis on clinical and ultrasonographicfeatures. Two specimens on gross examination showed mass lesions, and hence were suspected to becarcinoma of the gallbladder. Subsequent frozen sectionand histopathology demonstrated xanthogranulomatouscholecystitis. Only one case of xanthogranuloma was found to be associated with carcinoma of thegallbladder but no firm association could be establishedbetween xanthogranulomatous cholecystitis and carcinomaof the gallbladder.     

Genetic analysis of xanthogranulomatous cholecystitis: precancerous lesion of gallbladder cancer?

Xanthogranulomatous Cholecystitis is a chronic inflammatory disease of the gallbladder, a variant of the chronic cholecystitis. As xanthogranulomatous cholecystitis is occasionally seen with carcinoma of the gallbladder, the association with cancer is a controversial issue. A focal type of xanthogranulomatous cholecystitis is found simultaneously with gastric cancer diagnosed preoperatively. The resected specimen was genetically studied. Polymerase chain reaction amplification, single-strand conformational polymorphism analysis for mutation of p53 showed no abnormality indicating that less association with cancer in which the mutation of p53 is often seen. Etiopathologic factors of xanthogranulomatous cholecystitis might have relation with cancer, but xanthogranulomatous cholecystitis itself may not be the direct cause for cancer.     

A case of hepatitis C-associated osteosclerosis with xanthogranulomatous cholecystitis

A 62-year-old woman presented with a markedly increased serum ALP level of skeletal origin during a regular follow-up of chronic hepatitis C. Serum calcium, phosphorus, and intact-PTH levels were normal and bone turnover markers were increased. Her generalized bone density was diffusely increased. These findings were consistent with hepatitis C-associated osteosclerosis (HCAO). She underwent cholecystectomy, as gallbladder cancer was suspected; however, histopathological findings demonstrated xanthogranulomatous cholecystitis. After cholecystectomy, serum ALP level and bone turnover markers were gradually decreased. This may indicate the existence of a novel osteogenic factor in the gallbladder in HCAO.     

Extended surgical resection for xanthogranulomatous cholecystitis mimicking advanced gallbladder carcinoma： A case report and review of literature

Xanthogranulomatous cholecystitis (XGC) is a destructive inflammatory disease of the gallbladder, rarely involving adjacent organs and mimicking an advanced gallbladder carcinoma. The diagnosis is usually possible only after pathological examination. A 46 year-old woman was referred to our center for suspected gallbladder cancer involving the liver hilum, right liver lobe, right colonic flexure, and duodenum. Brushing cytology obtained by endoscopic retrograde cholangiography (ERC) showed high-grade dysplasia. The patient underwent an en-bloc resection of the mass, consisting of right lobectomy, right hemicolectomy, and a partial duodenal resection. Pathological examination unexpectedly revealed an XGC. Only six cases of extended surgical resections for XGC with direct involvement of adjacent organs have been reported so far. In these cases, given the possible coexistence of XGC with carcinoma, malignancy cannot be excluded, even after cytology and intraoperative frozen section investigation. In conclusion, due to the poor prognosis of gallbladder carcinoma on one side and possible complications deriving from highly aggressive inflammatory invasion of surrounding organs on the other side, it seems these cases should be treated as malignant tumors until proven otherwise. Clinicians should include XGC among the possible differential diagnoses of masses in liver hilum.     

Xanthogranulomatous choledochitis: a previously undescribed mass lesion of the hepatobiliary and ampullary region

Solid liver and pancreatic masses are commonly neoplastic in nature; however, inflammatory lesions mimicking carcinoma are at times encountered in these sites. We report two cases of previously undescribed inflammatory mass lesions of the liver and pancreas that originated in the biliary tract. Detailed clinical and histologic evaluations were performed in two patients who underwent right partial hepatic lobectomy and Whipple's resection for presumed hepatic and pancreatic neoplasms. In case 1, with a remote history of cholecystectomy and recent extraction of a stone from the common bile duct, a liver mass in segment 6 was discovered incidentally. In case 2, a periampullary pancreatic mass was diagnosed radiographically following papillotomy and stent insertion for stricture and biliary calculous disease. The histologic findings in both cases were similar, localized around a part of the biliary tract, and consisted of inspissated bile, acute and chronic inflammation, abundant lipid-laden macrophages, fibrosis, and giant cell reaction. No neoplasm was identified. On the basis of the close resemblance of these features to those seen in xanthogranulomatous cholecystitis, the lesions seen here were termed xanthogranulomatous choledochitis. In conclusion, xanthogranulomatous choledochitis is a benign inflammatory process involving the biliary tract that can form a mass lesion within the liver or pancreas and thus mimic a neoplasm. Extensive sampling of the lesion is required to rule out an underlying neoplastic process. In our patients a propensity to form lithogenic bile and a prior history of biliary tract operative procedure were present.     

Xanthogranulomatous cholecystitis mimicking gallbladder carcinoma with a false-positive result on fluorodeoxyglucose PET

Recently, several reports have demonstrated that fluorine-18 fluorodeoxyglucose positron emission tomography （FDG-PET） is useful in differentiating between benign and malignant lesions in the gallbladder. However, there is a limitation in the ability of FDG-PET to differentiate between inflammatory and malignant lesions. We herein present a case of xanthogranulomatous cholecystitis misdiagnosed as gallbladder carcinoma by ultrasonography and computed tomography. FDG-PET also showed increased activity. In this case, FDG-PET findings resulted in a false-positive for the diagnosis of gallbladder carcinoma.     

Suprahepatic gallbladder and right lobe anomaly of the liver in patients with biliary cancers

Summary The suprahepatic region is a rare ectopic location of the gallbladder. It usually combines with right lobe anomaly of the liver. Here we report two unusual cases of suprahepatic gallbladder with agenesis or hypogenesis of the right lobe of the liver and biliary cancer. A patient with a gallbladder tumor was admitted to our emergency room with acute cholecystitis and liver abscess. Imaging examinations and operation confirmed the suprahepatic position of gallbladder, agenesis of the right lobe, and dissemination of gallbladder cancer. In the patient with cholangiocarcinoma, CT scans and percutaneous transhepatic cholangiography documented the presence of a hilar tumor and hypogenesis of the right lobe. Both of these patients died from biliary tract cancer soon after operation.     

Kyste hydatique du foie ouvert dans la vésicule biliaire : à propos d’un cas

The opening in the intra hepatic bile ducts is the most common complication of hepatic hydatid cyst. The opening in the gallbladder is unusual.We report the case of a 65-year-old woman with complaints of right upper quadrant pain and fever. Various explorations have found a hydatid acute cholecystitis due to an opening of a hydatid cyst of the liver in the gallbladder. Treatment was surgical.     

Hypogenesis of right hepatic lobe accompanied by portal hypertension: Case report and review of 31 Japanese cases

We report a rare case of hypogenesis of the right lobe of the liver with portal hypertension and a review of 31 cases of agenesis or hypogenesis of the right hepatic lobe reported in Japan. A 74-year-old man consulted our hospital for further examination after a mass screening for gastric cancer. On physical examination liver enlargement was palpable, but liver function tests were normal. Abdominal ultrasonography, computed tomography, technetium-99m liver scintigraphy, and endoscopic retrograde cholangiopancreatography revealed a small right hepatic lobe and moderate splenomegaly, in contrast to a hypertrophic lateral segment of the left hepatic lobe, as well as ectopic dislocation of the gallbladder. Endoscopic examination revealed esophageal varices, indicating portal hypertension. Abdominal angiography demonstrated mild shunt flow between the hepatic artery feeding from the gastroduodenal artery and the portal vein. A biopsy specimen taken from both lobes of the liver showed normal liver tissue histologically. Based on these findings, we made a definite diagnosis of hypogenesis of the right lobe of the liver with portal hypertension. The present case appears to be the first such case accompanied by portal hypertension reported in Japan.     

Operative approach to gallbladder cancer

In most Western countries gallbladder cancer is a rare tumor with a poor prognosis. The majority of patients present with advanced-stage tumors (stage IV) that are not amenable to surgical resection. At the other end of the spectrum a small percentage of patients present with stage I disease that may be cured by cholecystectomy. The role for surgery in patients with stage II and III disease remains controversial, but most hepatobiliary surgeons believe that an aggressive surgical approach improves survival for these patients. However, the extent of hepatic and lymph node resection, the need for resection of the extrahepatic ducts in nonjaundiced patients, the role of vascular resection, and the advisability of hepatopancreatoduodenectomy remain a matter of debate. Although no data from prospective, randomized studies are available, resection of the gallbladder and adjacent liver with or without the extrahepatic bile ducts and with a regional lymph node dissection is the operative approach recommended for selected patients with gallbladder cancer.     

The role of lymphatic drainage of the liver in gallbladder cancer: a case report.

We report a case of a patient with a unique lymph node relapse after right hepatectomy and aggressive lymph node dissection for gallbladder cancer. There was extensive involvement of the hepatic parenchyma from the primary tumor, but no extension to the lymph nodes or other adjacent organs. Seventeen months later, the patient underwent re-dissection of the retroperitoneal lymph nodes with right nephrectomy and partial resection of the vena cava because of lymph node recurrence at the hilum of the right kidney. This pattern of lymph node metastasis to the right side of the vena cava from gallbladder cancer invading the liver is probably due to the distinct lymphatic drainage of the liver.     

Xanthogranulomatous cholecystitis]

Ten cases of xanthogranulomatous cholecystitis are presented, 5 women and 5 men, from a total of 439 cholecystectomies (2.2%). In 50% of cases the clinical course was consistent with acute cholecystitis; in 30%, gallbladder cancer was suspected preoperatively; and in 70% of cases cancer was suspected during surgery but intraoperative biopsies showed no malignancy. Definitive pathological findings included early carcinoma of the gallbladder in two patients, and a cholecystocolic fistula in one patient. A perforated gallbladder was found in one patient. The incidence of postoperative septic complications was 18.1%, a figure that doubles that of elective biliary surgery in our hospital.     

Hepatic artery pseudoaneurysm associated with cholecystitis that ruptured into the gallbladder

Pseudoaneurysm of the hepatic artery due to cholecystitis may be very rare, and in our survey of the literature, the present case report is the first case of such a pseudoaneurysm. A 64-year-old woman presented with upper gastrointestinal bleeding and severe epigastric pain. Upper gastrointestinal tract endoscopy revealed blood coming out of the papilla of Vater. Color-Doppler ultrasound imaging showed a pulsatile wave pattern in an echogenic lesion inside the gallbladder. Contrast-enhanced computed tomography demonstrated a 3-cm pseudoaneurysm in the distended gallbladder. Angiography disclosed extravasation originating from the right hepatic artery. Emergency selective transcatheter arterial embolization was performed, with intravascular stainless steel microcoils, and complete occlusion of the pseudoaneurysm was achieved. The patient underwent cholecystectomy with resection of the extrahepatic bile duct and biliary reconstruction in a Roux-en-Y fashion. Macroscopically, the resected gallbladder contained clotted blood and multiple cholesterol stones. Microscopically, the mucosa of the gallbladder showed extensive necrosis and many inflammatory cells. The final diagnosis was pseudoaneurysm of the hepatic artery associated with calculous gangrenous cholecystitis. Although the mechanism of the pseudoaneurysm remains speculative, severe inflammatory reaction in the gallbladder may have infiltrated the liver parenchyma and may have eroded the wall of the hepatic artery, thus forming a pseudoaneurysm. Hemobilia is one of the important differential diagnoses when unexplained gastrointestinal bleeding is observed, especially in patients with hepatobiliary diseases.     

Xanthogranulomatous Cholecystitis Masquerading as Gallbladder Carcinoma

We herein present a case of xanthogranulomatous cholecystitis which involved hoth the liver and transverse colon, clinically mimicking gallbladder carcinoma. Such cases may sometimes he judged inoperable due to extensive extra-gallbladder invasion, and thus it is necessary for physicians to take this lesion into consideration when making a diagnosis. An intraoperative biopsy is necessary, therefore, even when the feaures seem to clearly indicate inoperable carcinoma.     

Xanthogranulomatous cholecystitis. X-ray computed tomographic study of a pseudotumoral form

The authors report a case of xanthogranulomatous cholecystitis in a 63-year-old woman hospitalized because of a right hypochondrium mass. Ultrasonography, computed tomography, and celiac arteriogram showed a gallbladder tumour. Surgical management allowed subtotal tumour resection. Diagnosis of xanthogranulomatous cholecystitis was established by histological examination. Twenty-two months later, patient is in good health. From this observation and a world-wide literature review (90 previously reported cases), the authors analyse the clinical, pathological, and pathophysiological features of this very unusual entity.