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CASE REPORT |
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Year : 2013 | Volume
: 16
| Issue : 4 | Page : 168-170 |
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Xanthogranulomatous cholecystitis: Report of a case with coexistent adenocarcinoma
Sujata Jetley1, Safia Rana1, Usha Agrawal2, Zeeba S Jairajpuri1
1 Department of Pathology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi, India 2 National Institute of Pathology, ICMR, Safdurjung Hospital Campus, New Delhi, India
Date of Web Publication | 21-Jan-2014 |
Correspondence Address: Zeeba S Jairajpuri Department of Pathology, Hamdard Institute of Medical Sciences and Research, Hamdard Nagar, Jamia Hamdard, New Delhi - 110 062 India
Source of Support: None, Conflict of Interest: None | Check |
DOI: 10.4103/1118-8561.125569
Xanthogranulomatous cholecystitis (XGC) is an uncommon benign focal or diffuse destructive inflammatory disease of the gallbladder. Pre-operative and intra-operative diagnosis is difficult and it often mimics a gallbladder carcinoma (GBC). This diagnostic dilemma caused by equivocal imaging studies and intra-operative findings can at times be aggravated by a histological picture which mimics a neoplasm. Apart from occasionally presenting as an infiltrating mass lesion with adjacent organ invasion like a malignant neoplasm, XGC can also infrequently be associated with GBC. Lack of awareness that both XGC and carcinoma can co-exist as well as interpreter's inexperience may lead to a missed diagnosis. We present a case of to illustrate this coexistence and conclude that XGC and carcinoma of the gall bladder may co-exist and present a diagnostic dilemma. We recommend increased awareness of this knowledge among radiologists, surgeons and pathologists to raise its awareness. Keywords: adenocarcinoma, cholecystitis, xanthogranulomatous
How to cite this article: Jetley S, Rana S, Agrawal U, Jairajpuri ZS. Xanthogranulomatous cholecystitis: Report of a case with coexistent adenocarcinoma. Sahel Med J 2013;16:168-70 |
How to cite this URL: Jetley S, Rana S, Agrawal U, Jairajpuri ZS. Xanthogranulomatous cholecystitis: Report of a case with coexistent adenocarcinoma. Sahel Med J [serial online] 2013 [cited 2024 Mar 28];16:168-70. Available from: https://www.smjonline.org/text.asp?2013/16/4/168/125569 |
Introduction | | |
Xanthogranulomatous cholecystitis (XGC) is an uncommon focal or diffuse destructive inflammatory disease of the gallbladder which is considered to be a variant of conventional chronic cholecystitis. The lesion is characterized by distinct pathologic features on both gross and microscopic examination. [1],[2],[3] Pre-operative and intra-operative diagnosis is difficult and it often mimics a gallbladder carcinoma (GBC). XGC does not only occasionally present as an infiltrating mass lesion with adjacent organ invasion like a malignant neoplasm, it can also infrequently be associated with GBC. We present a case of coexisting simultaneous XGC and carcinoma of the gallbladder in a patient who underwent cholecystectomy for symptomatic gallstone disease with no pre-operative suspicion of malignancy.
Case Report | | |
A 65-year-old female patient presented with the complaints of recurrent pain in the right hypochondrium over the last 6 months. The patient was a known hypertensive, on medical management for the past 4-5 years and there was no past history of diabetes, tuberculosis or any previous surgery. General physical examination of the patient was unremarkable. There was no icterus and her blood pressure was well-controlled. Laboratory investigations revealed hemoglobin 12.4 g/dl, erythrocyte sedimentation rate, 22; total leucocyte count 8,600/mm 3 Differential Leukocyte count showed polymorphs 88%, lymphocytes 10% and eosinophils 02% respectively. Routine urine examination and microscopy was normal. Biochemical investigations showed fasting blood glucose of 110 mg/dl Liver and kidney function tests were within the normal limits. Ultrasonogram of the upper abdomen showed an enlarged gallbladder with a thickened wall and a pericholecystic collection. Single gallstone was present in the gallbladder lumen. The clinical impression was of symptomatic gall stone disease and the patient was taken up for laparoscopic cholecystectomy. Intra-operatively, a distended gallbladder with a single stone was seen. Dense adhesions between the gall bladder, the liver bed and omentum were also present. Gross examination showed a gallbladder measuring 6 cm × 2.5 cm with a congested external surface and single calculi in the lumen. Wall thickness ranged from 8 mm to 10 mm with multiple yellow brown streaks at places. No intraluminal growth or mass lesion was seen. Microscopic examination showed a well differentiated adenocarcinoma that infiltrated the full thickness of the muscular layer associated with a desmoplastic reaction. Spindle shaped xanthomatous cells with a storiform growth pattern were seen with cholesterol clefts, hemosiderin deposits and mononuclear cell infiltrates [Figure 1]. Other areas showed few ill-defined aggregates of xanthomatous cells closely related to the Rokitansky Aschoff sinuses. The resected margin of the gallbladder was free from tumor involvement. Immunohistochemical studies were carried out and staining with CD68 monoclonal antibody showed homogenous cytoplasmic staining of the infiltrating xanthoma cells [Figure 2]. Staining for epithelial membrane antigen (EMA) was done and found to be positive in the glands formed by the malignant epithelial cells [Figure 3]. | Figure 1: Microphotograph showing xanthogranulomatous foci composed of sheets of histiocytes, xanthomatous cells with numerous giant cells and cholesterol clefts (H and E, ×10)
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| Figure 2: Microphotograph showing CD68 positive histiocytes (immunohistochemistry, ×10)
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| Figure 3: Microphotograph showing epithelial membrane antigen positive malignant epithelium of the glands (immunohistochemistry, ×20)
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Discussion | | |
XGC is recognized as an uncommon form of chronic cholecystitis. Christensen and Ishak were among the first to describe this entity as a pseudotumor of the gallbladder (fibroxanthogranulomatous cholecystitis) with an unusual, destructive type of inflammation, desmoplasia, pericholecystic infiltration and hepatic involvement. [4] In 1981 the name XGC was proposed in a review of 40 cases from the Armed Forces institute of Pathology. [5] There is no information in previous literature about the overall incidence of XGC, although there are several reported small series Its clinical implications are that imaging studies and intra-operative appearances may confuse it with GBC and hence a correct histological diagnosis is critical to the further management of the patient. The presence of hypoechoic nodules or bands in the gallbladder wall is a characteristic sonographic finding in XGC. Cholelithiasis and a thickened gallbladder wall are also frequently seen, all of which show considerable overlap with sonographic findings in early GBC. [6] Similarly, intra-operative findings like adhesions with adjoining viscera with fistula formation are common and the presentation is often that of a gallbladder mass that mimics GBC. Poor visualization of the Calot's triangle due to the adhesions often results in an unsuccessful laparoscopic cholecystectomy with a high conversion rate to open cholecystectomy. This diagnostic dilemma caused by equivocal imaging studies and intra-operative findings can at times be aggravated by a histological picture which mimics a neoplasm. Spindly, elongated appearing xanthoma cells with a storiform growth pattern may be misinterpreted as malignancy by the novice. However, XGC alone lacks the unequivocal evidence of malignancy such as cellular pleomorphism and atypical mitosis. Nonetheless, the occasional coexistence of XGC and carcinoma of the gallbladder may present diagnostic confusion. Simultaneous XGC and GBC have been reported in some series with incidences ranging from 2% to 7.5% respectively [7],[8],[9] Benbow reterospectively reviewed 35 cases.of GBC reported at the Manchester Royal Infirmary and found associated XGC in 3 of the cases which had been hitherto overlooked. [5] Fine-needle aspiration cytology is considered to be an important pre-operative investigative tool to exclude coexisting malignancy, especially when the inflammation is localized to the gallbladder and imaging studies are suggestive of XGC. [1] Rastogi and co-workers also suggested intra-operative frozen section examination and immunostaining for markers such as CD68, EMA, CK in florid cases of XGC in which there was a suspicion of malignancy. [10] However, a rare presentation with no pre-operative suspicion of XGC and histology showing both XGC and GBC may occur as was seen in the present case.
Conclusion | | |
XGC and carcinoma of the gall bladder may co-exist and present a diagnostic dilemma. We recommend increased awareness of this knowledge among radiologists, surgeons and pathologists.
References | | |
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8. | Solanki RL, Arora HL, Gaur SK, Anand VK, Gupta R. Xanthogranulomatous cholecystitis (XGC): A clinicopathological study of 21 cases. Indian J Pathol Microbiol 1989;32:256-60. |
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10. | Rastogi A, Singh DK, Sakhuja P, Gondal R. Florid xanthogranulomatous cholecystitis masquerading as invasive gallbladder cancer leading to extensive surgical resection. Indian J Pathol Microbiol 2010;53:144-7. [PUBMED] |
[Figure 1], [Figure 2], [Figure 3]
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