Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 
Home Print this page Email this page
Users Online:: 191

 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 16  |  Issue : 4  |  Page : 168-170

Xanthogranulomatous cholecystitis: Report of a case with coexistent adenocarcinoma


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 Publication21-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
Login to access the Email id


DOI: 10.4103/1118-8561.125569

Rights and Permissions
  Abstract 

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 2022 Jan 22];16:168-70. Available from: https://www.smjonline.org/text.asp?2013/16/4/168/125569


  Introduction Top


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 Top


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)

Click here to view
Figure 2: Microphotograph showing CD68 positive histiocytes (immunohistochemistry, ×10)

Click here to view
Figure 3: Microphotograph showing epithelial membrane antigen positive malignant epithelium of the glands (immunohistochemistry, ×20)

Click here to view



  Discussion Top


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 Top


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 Top

1.Krishnani N, Shukla S, Jain M, Pandey R, Gupta RK. Fine needle aspiration cytology in xanthogranulomatous cholecystitis, gallbladder adenocarcinoma and coexistent lesions. Acta Cytol 2000;44:508-14.  Back to cited text no. 1
[PUBMED]    
2.Dixit VK, Prakash A, Gupta A, Pandey M, Gautam A, Kumar M, et al. Xanthogranulomatous cholecystitis. Dig Dis Sci 1998;43:940-2.  Back to cited text no. 2
[PUBMED]    
3.Roberts KM, Parsons MA. Xanthogranulomatous cholecystitis: Clinicopathological study of 13 cases. J Clin Pathol 1987;40:412-7.  Back to cited text no. 3
[PUBMED]    
4.Christensen AH, Ishak KG. Benign tumors and pseudotumors of the gallbladder. Report of 180 cases. Arch Pathol 1970;90:423-32.  Back to cited text no. 4
[PUBMED]    
5.Benbow EW. Xanthogranulomatous cholecystitis associated with carcinoma of the gallbladder. Postgrad Med J 1989;65:528-31.  Back to cited text no. 5
[PUBMED]    
6.Jetley S, Rana S, Khan S, Hassan MJ, Jairajpuri ZS. Incidental gall bladder carcinoma in laparoscopic cholecystectomy: A report of 6 cases and a review of the literature. J Clin Diagn Res 2013;7:85-8.  Back to cited text no. 6
    
7.Parra JA, Acinas O, Bueno J, Güezmes A, Fernández MA, Fariñas MC. Xanthogranulomatous cholecystitis: Clinical, sonographic, and CT findings in 26 patients. AJR Am J Roentgenol 2000;174:979-83.  Back to cited text no. 7
    
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.  Back to cited text no. 8
    
9.Guzmán-Valdivia G. Xanthogranulomatous cholecystitis: 15 years' experience. World J Surg 2004;28:254-7.  Back to cited text no. 9
    
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.  Back to cited text no. 10
[PUBMED]  Medknow Journal  


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
   Abstract
  Introduction
  Case Report
  Discussion
  Conclusion
   References
   Article Figures

 Article Access Statistics
    Viewed2355    
    Printed69    
    Emailed0    
    PDF Downloaded251    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]