Xanthogranulomatous cholecystitis (XGC) is an unusual and destructive inflammatory process that is characterized by thickening of the gallbladder (GB) wall with a tendency to adhere to neighboring organs. XGC is often mistaken for GB carcinoma, and the frequency of the coexistence of these two lesions is approximately 10%. Therefore, in case of severe XGC, there is chance of either overlooking the carcinoma or other significant lesions. CA 19-9 is commonly measured in the serum of patients with hepatobiliary malignancies. Although CA 19-9 can be elevated in benign conditions such as cholestasis, pancreatitis, tuberculosis, thyroid disease etc., malignancy should be considered at first in setting of its significant and persistent elevation. We report a case of a 62-year-old man who showed continuously rising level of CA19-9 over 2000 U/mL after cholecystectomy for xanthogranulomatous cholecystitis and finally was diagnosed as cholangiocarcinoma by short-term follow up.
Bile Duct Neoplasms/*diagnosis/pathology/radiography ; *Bile Ducts, Intrahepatic ; CA-19-9 Antigen/*blood ; Cholangiocarcinoma/*diagnosis/pathology/radiography ; Cholecystitis/pathology/*surgery ; Granuloma/pathology/*surgery ; Humans ; Male ; Middle Aged ; Positron-Emission Tomography ; Tomography, X-Ray Computed ; Xanthomatosis/pathology/*surgery

Xanthogranulomatous inflammation (XGI) is a rare, idiopathic process in which lipid-laden histiocytes are deposited at various locations in the body. Although XGI has been reported to occur in various organs such as the gallbladder, kidney, bone, stomach, colon, appendix, lymph nodes, urachus, and urinary bladder and in soft tissues, xanthogranulomatous pancreatitis (XGP) is extremely rare. Herein, we report a case of XGP occurring in a 70-yr-old woman, who presented with abdominal pain for several months. On physical examination, mild epigastric tenderness was noted. Abdomen CT scan revealed a low attenuated mass in uncinate process of pancreas, suggesting malignant lesion. Whipple's operation was performed and the final pathologic diagnosis was XGP. The patient's post-operative course was uneventful, and no recurrence was found within 7 months of the operation. When a pancreatic mass does not show clinico-radiological features typical of common pancreatic neoplasms, XGP should be considered for a differential diagnosis.
Aged ; Diagnosis, Differential ; Duodenum/surgery ; Female ; Granuloma/complications/*diagnosis/pathology ; Humans ; Pancreas/surgery ; Pancreatic Neoplasms/pathology ; Pancreatitis/complications/*diagnosis/pathology ; Positron-Emission Tomography ; Tomography, X-Ray Computed ; Xanthomatosis/complications/*diagnosis/pathology

There have been reports of the coexistence of abdominal aortic aneurysm (AAA) with intra-abdominal malignancy including gastric, colonic, pancreatic, and renal. We herein report a case of a previously undiagnosed AAA and a presenting complaint consistent with acute cholecystitis. Following cholecystectomy, this was noted to be a rare form of chronic cholecystitis: xanthogranulomatous cholecystitis. There is a known possible association of this uncommon condition with gallbladder cancer. The management of concomitant pathologies can present a real challenge to the multidisciplinary team, especially with large aneurysms.
Adenocarcinoma/*complications/radiography/secondary/surgery ; Aged ; Aortic Aneurysm, Abdominal/*complications/radiography/surgery ; Biopsy ; Blood Vessel Prosthesis Implantation ; Cholecystectomy ; Cholecystitis/*complications/pathology/radiography/surgery ; Endovascular Procedures ; Female ; Gallbladder Neoplasms/*complications/pathology/radiography/surgery ; Granuloma/*complications/pathology/radiography/surgery ; Humans ; Tomography, X-Ray Computed ; Treatment Outcome ; Xanthomatosis/*complications/pathology/radiography/surgery

Xanthogranulomatous lesion is a rare condition in which lipid-laden histiocytes are deposited at various locations in the body. Xanthogranulomatous pancreatitis (XGP) associated with an intraductal papillary mucinous tumor (IPMT) is extremely rare. In this study, we described a case of XGP associated with IPMT and include a review of the literature. A pancreatic cystic mass was detected in a 72-yr-old woman by abdominal computed tomography. Pylorus-preserving pancreaticoduodenectomy was performed and diagnosis of XGP combined with intraductal papillary mucinous carcinoma in situ was made. After 13 months of follow-up, the patient is in good health without any evidence of tumor recurrence. Although XGP associated with IPMT is rare, we suggest that such cases should be brought to the attention of clinical investigators, as it may produce clinical features that mimic pancreatic cancer.
Adenocarcinoma, Mucinous/*diagnosis/pathology/surgery ; Aged ; Carcinoma in Situ/*diagnosis/pathology/surgery ; Carcinoma, Pancreatic Ductal/*diagnosis/pathology/surgery ; Carcinoma, Papillary/*diagnosis/pathology/surgery ; Diagnosis, Differential ; Female ; Granuloma/complications/*diagnosis ; Humans ; Magnetic Resonance Imaging ; Pancreatic Neoplasms/*diagnosis/pathology/surgery ; Pancreaticoduodenectomy ; Pancreatitis/complications/*diagnosis ; Tomography, X-Ray Computed ; Xanthomatosis/complications/*diagnosis

Xanthogranulomatous cholecystitis (XGC) is a rare inflammatory disease of the gallbladder. Not only does XGC occasionally present as a mass formation with adjacent organ invasion like a malignant neoplasm, it can also infrequently be associated with gallbladder cancer. In the situation, it is difficult to make a differential diagnosis between the diseases. Here, we describe a case of a simultaneous XGC and a carcinoma of the gallbladder in a 61-year-old woman. To the best of our knowledge, there are only a small number of reports on this combination of diseases.
Adenocarcinoma/complications/diagnosis/*pathology/surgery ; Biopsy, Needle ; Cholangiopancreatography, Endoscopic Retrograde ; Cholecystitis/complications/diagnosis/*pathology/surgery ; Endosonography ; Female ; Gallbladder Neoplasms/complications/diagnosis/*pathology/surgery ; Granuloma/complications/diagnosis/*pathology ; Humans ; Immunohistochemistry ; Middle Aged ; Neoplasm Staging ; Prognosis ; Risk Assessment ; Tomography, X-Ray Computed ; Xanthomatosis/complications/diagnosis/*pathology

Actins ; metabolism ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Diagnosis, Differential ; Female ; Granuloma, Plasma Cell ; metabolism ; pathology ; Histiocytoma, Benign Fibrous ; diagnostic imaging ; metabolism ; pathology ; surgery ; Humans ; Lung Neoplasms ; diagnostic imaging ; metabolism ; pathology ; surgery ; Middle Aged ; Neprilysin ; metabolism ; Pneumonectomy ; methods ; Radiography ; Sarcoma ; metabolism ; pathology ; Solitary Fibrous Tumors ; metabolism ; pathology ; Vimentin ; metabolism ; Xanthomatosis ; metabolism ; pathology

OBJECTIVETo study the clinicopathologic characteristics of atypical fibrous histiocytoma (AFH), with emphasis on differential diagnosis.

METHODSThe clinical and pathologic features were reviewed in 24 cases of AFH (from 2007 to 2012). The follow-up data were analyzed. Immunohistochemical study using EnVision method was carried out.

RESULTSThere were 10 males and 14 females with age at presentation ranging from 8 to 67 years (mean = 41 years and median = 39 years). The tumor occurred in the extremities (number = 14), trunk (number = 8) or head and neck region (number = 2). Apart from one case, all were located in the dermis. The clinical appearance was similar to those of classic fibrous histiocytoma. Histologically, the tumor was characterized by various number of hyperchromatic bizarre cells scattered in the background. Mitotic figures including atypical ones were noted, especially in the more cellular areas. Immunohistochemical study showed that the tumor cells expressed vimentin. Focal positivity for alpha-smooth muscle actin was demonstrated in some cases. Follow-up information was available in 14 cases. Three of them suffered local recurrence. None of these cases had distant metastasis.

CONCLUSIONSAtypical fibrous histiocytoma represents a pleomorphic variant of fibrous histiocytoma. Although the tumor exhibits worrisome features, it usually pursuits a relatively benign course. Nevertheless, rare cases may recur, especially after incomplete excision. AFH is sometimes mistaken as atypical fibroxanthoma. A distinction between the two entities is warranted as they represent two different entities.

Actins ; metabolism ; Adolescent ; Adult ; Aged ; Back ; pathology ; Child ; Diagnosis, Differential ; Extremities ; pathology ; Female ; Follow-Up Studies ; Histiocytoma, Benign Fibrous ; metabolism ; pathology ; surgery ; Humans ; Male ; Middle Aged ; Neoplasm Recurrence, Local ; Retrospective Studies ; Skin Neoplasms ; metabolism ; pathology ; surgery ; Vimentin ; metabolism ; Xanthomatosis ; pathology ; Young Adult