Cystic lymphangioma of the jejunal mesentery in an adult： A case report

We herein describe the case of a 27-year-old female, who presented with a large mass of the upper left abdominal cavity discovered incidentally, through an annual health examination. Preoperative studies including abdominal ultrasonography and magnetic resonance imaging were performed, but they could not accurately determine the nature of the tumor. At laparotomy, a large cystic tumor of the small bowel mesentery was found. Histopathologic examination diagnosed the tumor as a cystic lymphangioma. Although lymphangiomas are rare, especially in the abdomen of adults, they may sometimes present as acute abdomen, causing complications that require emergent surgery.     

Adult intussusception caused by cystic lymphangioma of the colon： A rare case report

We experienced a case of intussusception caused by cystic lymphangioma of the colon in a 32 years old female who was admitted to our hospital for the chief complaint of bloody stool. In the colonoscopic examination, cystic mass with stalk which had smooth mucosal surface was noted at the descending colon. Abdominal ultrasonography and computed tomography revealed left colon intussusception with a multilocular cystic tumor as a leading point. Emergent operation was performed. On the histopathologic examination, the cystically dilated spaces lined by endothelium and septated by fibrous septa were present. The pathological diagnosis was cystic lymphangioma of the colon. Although intussusception due to lymphangioma in an adult are rare, it should be taken into consideration that it is possible diagnosis.     

Spontaneous necrosis of solid gallbladder adenocarcinoma accompanied with pancreaticobiliary maljunction

A 71-year-old Japanese man with acute cholecystitis and an incarcerated gallbladder （GB） stone was admitted. Plain ultrasonography （US） incidentally detected a mass-like lesion in the fundus. Doppler US revealed that this elevated lesion had no blood flow. Computed tomography showed a relatively low-density mass, measuring 5 cm x 4 cm in diameter, with no positive enhancement. Magnetic resonance imaging showed a mass in the fundus with a slightly low intensity on Tl-weighted images and a slightly high intensity on T2-weighted images. We were agonized in making the qualitative diagnosis of mass-like lesions of the fundus, such as a benign tumor, cancer, or debris. We performed laparoscopic cholecystectomy, because the incarcerated GB stone clearly caused acute cholecystitis. Intra-operative cholangiography clearly revealed pancreaticobiliary maljunction. Amylase levels in the common bile duct and gallbladder were quite high. The elevated lesion in the fundus dearly showed severe necrosis. Although this necrotic nodule included non-viable adenocarcinoma cells, viable cancer cell nests were located in the muscularis propria and subcutaneous layer. Histopathological examination confirmed a solid adenocarcinoma. Thus, we diagnosed it as a gallbladder cancer, based on histopathological analysis of the resected specimen. We therefore undertook radical surgery, including wedge resection of the liver, radical dissection of regional lymph nodes, and resection of the extrahepatic bile duct. Histopathological findings revealed no cancer, hyperplasia or dysplasia in the additionally resected specimens. The patient was finally staged as T2, N0, H0, P0, M（-）, stage Ⅱ. We present the first case of spontaneous necrosis of solid gallbladder adenocarcinoma, with a review of previous studies.     

Obstructive jaundice and melena caused by hemocholecyst: A case report

A hemocholecyst (HC) is a clot-filled gallbladder caused by bleeding into its lumen. Obstructive jaundice caused by the compression of HC to the hilar biliary tract is likely to be misdiagnosed as cholangiocarcinoma and is extremely rare. We herein report a case of obstructive jaundice and melena caused by HC. A 57-year-old male patient presented with right upper quadrant pain associated with icteric sclera and melena was suspiciously diagnosed as having malignant cholangiocarcinmoa by abdominal ultrasonography, computed tomography and magnetic resonance imaging. Laparotomy found a hematoma in the gallbladder. The hematoma spread to the left hepatic lobe forming an exogenous mass which compressed the hilar biliary tract. Radical cholecystectomy and bile duct exploration with T-tube drainage were performed. Histopathological examination revealed massive necrosis of the gallbladder mucosa with inflammatory cells infiltration as well as intraluminal hematoma formation. One month after operation, a T-tube cholangiography revealed a normal biliary tree. We suggest that HC should be considered in patients with obstructive jaundice and melena after common causes are ruled out.     

Primary liposarcoma of gallbladder diagnosed by preoperative imagings： A case report and review of literature

A 49-year-old Japanese woman was referred to our department because of high fever and a huge abdominal mass. Computed tomography (CT) and magnetic resonance (MR) imagings revealed a tumor, about 30 cm in diameter, occupied the right hepatic lobe and the peritoneal cavity. Abdominal angiography showed that the tumor was fed mainly by the cystic artery. We pre-operatively diagnosed angiosarcoma of the gallbladder and performed tumor resection with cholecystectomy because the tumor was almost casplated, however the posterior wall of the gallbladder attached to the tumor firmly. Histologically, the tumor was composed of spindle cells including lipoblasts with cellular pleomorphism, which were also detected in the muscular layer of the gallbladder. We finally diagnosed pleomorphic liposarco-ma of the gallbladder. At 10 mo and 29 mo after the first operation, she underwent two more operations because of recurrence. Now she has a good quality of life 3 years and 6 mo after the first operation.     

Cystic micropapillary neoplasm of peribiliary glands with concomitant perihilar cholangiocarcinoma

We report a case of a 75-year-old man with cystic micropapillary neoplasm of peribiliary glands detected preoperatively by radiologic examination. Enhanced computed tomography showed a low-density mass 2.2 cm in diameter in the right hepatic hilum and a cystic lesion around the common hepatic duct. cholangiocarcinoma, right hepatectomy with caudate lobectomy and bile duct resection were performed. Pathological examination revealed perihilar cholangiocarcinoma mainly involving the right hepatic duct. The cystic lesion was multilocular and covered by columnar lining epithelia exhibiting increased proliferative activity and p53 nuclear expression; it also contained foci of micropapillary and glandular proliferation. Therefore, the lesion was diagnosed as a cystic micropapillary neoplasm of peribiliary glands and resembled flat branch-type intraductal papillary mucinous neoplasm of the pancreas. Histological examination showed the lesion was discontinuous with the perihilar cholangiocarcinoma. Immunohistochemistry showed the cystic neoplasm was strongly positive for MUC6 and that the cholangiocarcinoma was strongly positive for MUC5 AC and S100 P. These results suggest these two lesions have different origins. This case warrants further study on whether this type of neoplasm is associated with concomitant cholangiocarcinoma as observed in pancreatic intraductal papillary mucinous neoplasm with concomitant pancreatic duct adenocarcinoma.     

Cavernous mesenteric lymphangiomatosis mimicking metastasis in a patient with rectal cancer： A case report

Lymphangioma usually occurs in children and usually involves the skin. Mesenteric lymphangioma is extremely rare in adults. Typically, lymphangioma appears on computed tomography （CT） as a lower attenuation of a cystic mass, however, some cases appear to be a solid mass. We describe the CT and 18FFDG positron emission tomography/CT appearance in a case of jejunal and mesenteric cavernous lymphangiomatosis mimicking metastasis in an adult patient with rectal cancer.     

Metachronous bile duct cancer nine years after resection of gallbladder cancer

We report a rare case of a 74-year-old man with metachronous gallbladder cancer and bile duct cancer who underwent curative resection twice, with the operations nine years apart. At the age of 65 years, the patient underwent a cholecystectomy and resection of the liver bed for gallbladder cancer. This was a welldifferentiated adenocarcinoma, with negative resection margins （T2NOM0, stage Ⅰ B）. Nine years later, during a follow-up examination, abdominal computed tomography and MRCP showed an enhanced 1.7 cm mass in the hilum that extended to the second branch of the right intrahepatic bile duct. We diagnosed this lesion as a perihilar bile duct cancer, Bismuth type Ⅲ a, and performed bile duct excision, right hepatic Iobectomy and Roux-en-Y hepaticojejunostomy. The histological diagnosis was a well-differentiated adenocarcinoma with one regional lymph node metastasis （TINIM0, stage Ⅱ B）. Twelve months after the second operation, the patient is well, with no signs of recurrence. This case is compared with 11 other cases of metachronous biliary tract cancer published in the world medical literature.     

Anatomical variations of the cystic duct： Two case reports

Anatomical variations of the cystic duct often occur and may be encountered during cholecystectomy. Knowledge of the variable anatomy of the cystic duct and cysticohepatic junction is important to avoid significant ductal injury in biliary surgery. Here, we present two unusual cases with an anomalous cystic duct, namely, low lateral insertion and narrow-winding of the cystic duct. The first case was a 64-year-old man with cholelithiasis and chronic cholecystitis. During surgery, the entrance of the cystic duct was misidentified as being short and leading into the right hepatic duct. Further exploration showed multiple calculi in the right and common hepatic ducts. Cholecystectomy was completed, followed by T-tube drainage of the common and right hepatic ducts. Postoperative T-tube cholangiography demonstrated that the two T tubes were respectively located in the cystic and common hepatic duct. Six weeks later, the retained stones in the distal choledochus were extracted by cholangioscopy through the sinus tract of the T-tube. The second case was a 41-year-old woman, in which, preoperative endoscopic retrograde cholangiopancreatography （ERCP） revealed a long cystic duct, with a narrow and curved-in lumen. The patient underwent open cholecystectomy. Both patients were cured. The authors propose that preoperative ERCP or magnetic resonance cholangiopancreatography （MRCP）, and intraoperative cholangiography or cholangioscopy constitute a useful and safe procedure for determining anatomical variations of the cystic duct.     

Morphologic factors of biliary trees are associated with gallstone-related biliary events

AIM: To determine the risk factors for gallstone-related biliary events.METHODS: This retrospective cohort study evaluated magnetic resonance cholangiopancreatography imagesfrom 141 symptomatic and 39 asymptomatic gallstone patients who presented at a single tertiary hospital between January 2005 and December 2012.RESULTS: Logistic regression analysis showed significant differences between symptomatic and asymptomatic patients with gallstones in relation to the number of gallstones,the angle between the long axis of the gallbladder and the cystic duct,and the cystic duct diameter.Multivariate analysis showed that the number of gallstones(OR = 1.27,95%CI: 1.03-1.57; P = 0.026),the angle between the long axis of the gallbladder and the cystic duct(OR = 1.02,95%CI: 1.00-1.03; P = 0.015),and the diameter of the cystic duct(OR = 0.819,95%CI: 0.69-0.97; P = 0.018) were significantly associated with biliary events.The incidence of biliary events was significantly elevated in patients who had the presence of more than two gallstones,an angle of 92° between the gallbladder and the cystic duct,and a cystic duct diameter 6 mm.CONCLUSION: These findings will help guide the treatment of patients with asymptomatic gallstones.Clinicians should closely monitor patients with asymptomatic gallstones who exhibit these characteristics.     

Cystic lymphangioma of the gall-bladder: A case report

Intra-abdominal cystic lymphangiomas are rare lesions that can be difficult to diagnose. We present a report of a patient with a giant multilocular cystic lesion in the abdomen. Ultrasonography and computed tomography scans of the abdomen revealed that the cyst had originated in the gallbladder fossa. There was some calcification and thickening of the cyst wall. Endoscopic retrograde cholangiopancreatography demonstrated a medially deviated common bile duct, an elongated cystic duct and an inferior compressed gallbladder. There was no apparent communication between the cyst and the biliary tract; however, an abdominal angiogram revealed that the lesion was supplied by a branch of the cystic artery. Histological findings obtained intra-operatively were consistent with a cystic lymphangioma. Its characteristic histology was observed in the subserous layer of the gall-bladder. This case is a rare instance of a cystic lymphangioma originating in the gall-bladder.     

Gallbladder lymphangioma: a case report and review of the literature

Lymphangiomas are benign neoplasms usually occurring in childhood and located in the head and neck. Intraabdominal lymphangiomas account for less than 5% of cases. The involvement of the gallbladder is rare. We report a case of a 29-year-old woman who presented with right upper quadrant pain that had persisted for 6 months. Imaging with ultrasound and magnetic resonance imaging (MRI) revealed a multiseptated lesion surrounding the gallbladder. The patient underwent an exploratory laporatomy, and the mass was resected en bloc with the gallbladder. Histological evaluation of the cystic mass revealed findings consistent with lymphangioma. The prognosis is generally good after complete surgical excision, as was the case for our patient.     

Preoperative Diagnosis of Gallbladder Torsion by Magnetic Resonance Cholangiopancreatography

The patient was a 78-year-old woman who was diagnosed as having gallbladder torsion preoperatively. This is the first reported case diagnosed by magnetic resonance cholangiopancreatography (MRCP). Signs and symptoms of this condition are often subtle. Radiologic evaluation by ultrasonography and computed tomography (CT) showed acute cholecystitis with stone. Drip-infusion cholangiography CT failed to outline the gallbladder, and distortion of the extrahepatic bile ducts and interruption of the cystic duct were observed. MRCP showed 1) a v-shaped distortion of the extrahepatic bile ducts due to traction by the cystic duct, 2) tapering and twisting interruption of the cystic duct, 3) a distended and enlarged gallbladder that was deviated to the midline of the abdomen, and 4) a difference in intensity between the gallbladder and the extrahepatic bile ducts and the cystic duct. A definitive diagnosis of gallbladder torsion (volvulus) was made by MRCP preoperatively. If treated surgically, gallbladder detorsion before cholecystectomy is a helpful technique to avoid bile duct injury. This condition should be suspected in elderly women with acute cholecystitis or acute abdominal pain of unknown origin, and MRCP may be very useful in making a definitive diagnosis.     

Preoperative diagnosis of gallbladder torsion by magnetic resonance cholangiopancreatography

The patient was a 78-year-old woman who was diagnosed as having gallbladder torsion preoperatively. This is the first reported case diagnosed by magnetic resonance cholangiopancreatography (MRCP). Signs and symptoms of this condition are often subtle. Radiologic evaluation by ultrasonography and computed tomography (CT) showed acute cholecystitis with stone. Drip-infusion cholangiography CT failed to outline the gallbladder, and distortion of the extrahepatic bile ducts and interruption of the cystic duct were observed. MRCP showed 1) a v-shaped distortion of the extrahepatic bile ducts due to traction by the cystic duct, 2) tapering and twisting interruption of the cystic duct, 3) a distended and enlarged gallbladder that was deviated to the midline of the abdomen, and 4) a difference in intensity between the gallbladder and the extrahepatic bile ducts and the cystic duct. A definitive diagnosis of gallbladder torsion (volvulus) was made by MRCP preoperatively. If treated surgically, gallbladder detorsion before cholecystectomy is a helpful technique to avoid bile duct injury. This condition should be suspected in elderly women with acute cholecystitis or acute abdominal pain of unknown origin, and MRCP may be very useful in making a definitive diagnosis.     

Double cancer of the cystic duct and gallbladder associated with low junction of the cystic duct

We report a case of double cancer of the cystic duct and gallbladder associated with low junction of the cystic duct. A 73-year-old woman was admitted to the hospital complaining of upper abdominal pain. Endoscopic retrograde cholangiography showed a stenotic lesion in the lower common bile duct and no visualization of the cystic duct or gallbladder. Enhanced computed tomography revealed a heterogeneously enhanced tumorous lesion around the lower bile duct in the pancreatic head. A diagnosis of cancer arising from the cystic duct that entered the lower part of the common hepatic duct was made by intraductal ultrasonography, which showed an intraluminal protruding lesion in the cystic duct. Isolated gallbladder cancer was also diagnosed, by abdominal computed tomography. She underwent pancreaticoduodenectomy with dissection of regional lymph nodes. Histological examination revealed moderately differentiated adenocarcinoma of the cystic duct and well-differentiated adenocarcinoma of the gallbladder. Double cancer of the cystic duct and gallbladder is extremely rare, and this case also suggests a relationship between a low junction of the cystic duct and neoplasm in the biliary tract.     

Multiple carcinoid tumors of the gallbladder and cystic duct

A very rare case of multiple carcinoid tumors of the gallbladder and cystic duct is reported in a 77-year-old woman. Cholecystectomy and resection of the common bile duct, with regional lymph node dissection, were performed. The tumors consisted of a large mass (30×20×15mm) in the neck of the gallbladder and three small lesions in the cystic duct. Histologically, the tumor cells were arranged in solid nests, and showed many mitoses. Histochemical studies revealed argyrophilic and argentaffinic staining. Immunohistochemically, the tumor cells were positive for serotonin, chromogranin A, Leu 7, synaptophysin, and neuron-specific enolase. Thus, the present case was diagnosed as midguttype carcinoids.     

Floating gallbladder associated with histologically distinct double cancers

A case of unusually hypermobile floating gall-bladder in a 79-year-old woman with histologically distinct double cancers of the gallbladder is described. The patient presented with an abdominal cystic mass, which was palpable with easy mobility from the right lower quadrant practically to the left upper quadrant. Exploratory laparotomy was performed and the cystic mass was found to be a floating gallbladder. The cystic duct was elongated and obstructed, and had a long mesentery. After the operation, latent double cancers of the gallbladder were discovered on histopathological examination. The obstruction of the cystic duct was due to chronic inflammation and had resulted in hydrops of the gallbladder. This was suspected to have played an important role in the carcinogenesis. We believe that this is the first report of a floating gallbladder associated with double gallbladder cancers.     

A case of early cystic duct carcinoma concomitant with xanthogranulomatous cholecystitis (XGC)

We reported a case of early cystic duct carcinoma concomitant with xanthogranulomatous cholecystitis (XGC). This case was a 72-year-old man in whom thickening of the gallbladder wall was pointed out an abdominal ultrasonography and elevation of the CA19-9 level was detected at a local clinic. Endoscopic ultrasonography and CT demonstrated a mass in the cystic duct. Mapping biopsy using peroral cholangioscopy (POCS) revealed a diagnosis of cystic carcinoma with superficial flat growth, therefore a pylorus-preserving pancreatoduodenectomy was performed. Histopathological diagnosis was well differentiated papillotubular adenocarcinoma with superficial flat spread and the thickening of the gallbladder wall was XGC. A case of early cystic duct carcinoma concomitant with XGC is extremely rare.     

An extrahepatic bile duct metastasis from a gallbladder cancer mimicking Mirizzi's syndrome

We report a case of an extrahepatic bile duct metastasis from a gallbladder cancer that mimicked Mirizzi's syndrome on cholangiography. A 67-yr-old woman was admitted to our hospital with a diagnosis of acute calculous cholecystitis. As obstructive jaundice developed after the admission, percutaneous transhepatic biliary drainage was performed to ameliorate the jaundice and to evaluate the biliary system. Tube cholangiography revealed bile duct obstruction at the hepatic hilus, and extrinsic compression of the lateral aspect of the common hepatic duct, with nonvisualization of the gallbladder. No impacted cystic duct stone was visualized on CT or ultrasonography. Laparotomy revealed a gallbladder tumor as well as an extrahepatic bile duct tumor. We diagnosed that the latter was a metastasis from the gallbladder cancer, based on the histopathological features. This case is unique in that the extrahepatic bile duct metastasis obstructed both the common hepatic duct and the cystic duct, giving the appearance of Mirizzi's syndrome on cholangiography. Metastatic bile duct tumors that mimic Mirizzi's syndrome have not been previously reported. The presence of this condition should be suspected in patients with the cholangiographic features of Mirizzi's syndrome, when the CT or ultrasonographic findings fail to demonstrate an impacted cystic duct stone.