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CASE REPORT
Year : 2017  |  Volume : 31  |  Issue : 3  |  Page : 208-210

Xanthogranulomatous appendicitis: A rare histopathological entity


1 Department of Surgery, Regional Institute of Medical Sciences, Imphal, Manipur, India
2 Department of Pathology, JNIMS, Imphal, Manipur, India

Date of Web Publication17-Aug-2017

Correspondence Address:
Angelica Laiphrakpam
Regional Institute of Medical Sciences Doctors Colony, Quarters No. v/4, Lamphelpat - 795 004, Imphal, Manipur
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jms.jms_16_17

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  Abstract 


Xanthogranulomatous inflammation is a chronic inflammatory process most commonly described in the kidney and gallbladder. There are few reports in literature of this process occurring in the vermiform appendix. In this case, a 36-year-old male patient presented with typical signs and symptoms of acute appendicitis necessitating an emergency appendectomy. Histopathological examination revealed typical features of xanthogranulomatous inflammation in the appendix. We report this case in view of the rarity of its occurrence.

Keywords: Appendectomy, vermiform appendix, xanthogranulomatous inflammation


How to cite this article:
Laiphrakpam A, Singh KL, Devi LS, Moirangthem G S. Xanthogranulomatous appendicitis: A rare histopathological entity. J Med Soc 2017;31:208-10

How to cite this URL:
Laiphrakpam A, Singh KL, Devi LS, Moirangthem G S. Xanthogranulomatous appendicitis: A rare histopathological entity. J Med Soc [serial online] 2017 [cited 2019 Oct 20];31:208-10. Available from: http://www.jmedsoc.org/text.asp?2017/31/3/208/211095




  Introduction Top


Although acute appendicitis is one of the most common surgical conditions, xanthogranulomatous appendicitis is uncommon. It is a rare type of chronic inflammation leading to tissue destruction and localized proliferation of lipid-laden macrophages which is the characteristic histological feature with only a few reported cases. Here, we report a case of xanthogranulomatous appendicitis presenting as acute appendicitis necessitating appendicectomy. We also reviewed the limited medical literature.


  Case Report Top


A 36-year-old male patient presented to the emergency department with a history of pain in the right iliac fossa along with fever and vomiting for 1 day before admission. He had no history of any medical or surgical illness in the past. His family history was unremarkable. On physical examination, the patient was febrile. There was marked tenderness and rebound tenderness in the right iliac fossa. His complete hemogram, liver function tests, and kidney function tests were all within normal limits. Ultrasound scan of the abdomen revealed an appendix with a diameter of 8 mm with minimal periappendicular fluid collection with no faecolith. A computed tomography of abdomen was not done in this case.

The patient was taken for an emergency appendectomy. Intraoperatively, the appendix was found inflamed with the tip adherent to the surrounding bowel, but the base was healthy. There was minimal fluid collection. There was no faecolith and the lumen was patent.

His surgery was uneventful. Histopathological analysis of the appendix specimen showed the presence of numerous foamy histiocytes in a background of lymphocytes, neutrophils, plasma cells, and a few multinucleated giant cells [Figure 1] and [Figure 2]. No acid-fast bacilli was seen on ZN stain. Von Kossa and Pearl's stain was also done to rule out malakoplakia, but immunohistochemistry was not done. A diagnosis of xanthogranulomatous appendicitis was thus obtained.
Figure 1: H and E, ×10

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Figure 2: H and E, ×40

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His postoperative stay was uneventful. He was discharged on the 5th postoperative day, subsequent follow-up visits in the outpatient department at 2 weeks and 4 weeks were unremarkable.


  Discussion Top


Xanthogranulomatous inflammation is a form of chronic inflammation, characterized by the presence of lipid-laden macrophages admixed with lymphocytes, plasma cells, neutrophils, and often multinucleated giant cells with or without cholesterol clefts.[1] Any organ of the body can be involved in the disease process with the kidney and gallbladder being the most common.[2]

Osterlind[3] first described it in the kidney in 1944. Other organs showing xanthogranulomatous inflammation include endometrium, epididymis, fallopian tubes, bone, skin, appendix, urinary bladder, thyroid, and adrenal glands.[2],[4] It is rare in the appendix with only 12 known cases reported in literature.[5]

The exact etiopathogenesis is unknown. Proposed hypotheses include defective lipid transport, immunological disorders such as disturbed chemotaxis of polymorphonuclear cells and macrophages, a specific immune response toward Proteus and Escherichia infections, and lymphatic obstruction.[6],[7]

Microscopic examination of xanthogranulomatous inflammation usually reveals a nodular or diffuse collection of foamy histiocytes, intermixed with varying amounts of other inflammatory cells, such as multinucleated giant cells, lymphocytes, plasma cells, and eosinophils, as well as fibrosis. Occasionally, cholesterol clefts, granulation tissue, and necrotic debris are observed with reactive lymphoid hyperplasia.[8]

Guo and Greenson[9] reviewed appendectomies over a period of 4 years. They found that xanthogranulomatous inflammation occurred in 8 out of 22 (36.4%) of interval appendectomies but did not occur in emergency appendectomies group. They concluded that xanthogranulomatous inflammation is characteristically associated with chronic inflammation, with the presence of granulomas, xanthomatous cells, mural fibrosis, and transmural chronic inflammatory infiltrates. These changes may mimic chronic inflammation associated with Crohn's disease.

Kochhar et al.[10] reported a case of appendicular lump which was initially managed conservatively and failed. The patient underwent right hemicolectomy after developing intestinal obstruction. Finally, the patient went into septicemia and expired. Histopathological study showed features of xanthogranulomatous appendicitis.

In another case reported by Chuang et al.,[7] a 39-year-old patient underwent right hemicolectomy for a hard lump with suspicion of right colonic growth. Final histology revealed features of xanthogranulomatous appendicitis thereby showing that this phenomenon may simulate malignancy of the right colon. We presume that our finding of xanthogranulomatous inflammation in this acute setting is an acute recurrent attack of appendicitis with subclinical past attacks.


  Conclusion Top


Xanthogranulomatous inflammation is an uncommon healing pattern of appendicitis. It may be encountered in interval appendectomy specimens and the acute setting as seen in our case. It may mimic malignancy of the right colon leading to a much more extensive surgery than an appendectomy. It is pertinent to rule out other conditions by histopathological examination which may present with similar histology such as Crohn's disease and malakoplakia.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Cozzutto C, Carbone A. The xanthogranulomatous process. Xanthogranulomatous inflammation. Pathol Res Pract 1988;183:395-402.  Back to cited text no. 1
    
2.
Oh YH, Seong SS, Jang KS, Chung YW, Paik CH, Park YW, et al. Xanthogranulomatous inflammation presenting as a submucosal mass of the sigmoid colon. Pathol Int 2005;55:440-4.  Back to cited text no. 2
    
3.
Osterlind S. “Uber pyelonephritis xanthomatosa”. Acta Chir Scand 1944;90:369-76.  Back to cited text no. 3
    
4.
Gray Y, Libbey NP. Xanthogranulomatous salpingitis and oophoritis: A case report and review of the literature. Arch Pathol Lab Med 2001;125:260-3.  Back to cited text no. 4
    
5.
Jusoh AC, Ghani SA. Xathogranulomatous lesion in recurrent appendicitis. Formos J Surg 2016;49:114-8.  Back to cited text no. 5
    
6.
Antonakopoulos GN, Chapple CR, Newman J, Crocker J, Tudway DC, O'Brien JM, et al. Xanthogranulomatous pyelonephritis. A reappraisal and immunohistochemical study. Arch Pathol Lab Med 1988;112:275-81.  Back to cited text no. 6
    
7.
Chuang YF, Cheng TI, Soong TC, Tsou MH. Xanthogranulomatous appendicitis. J Formos Med Assoc 2005;104:752-4.  Back to cited text no. 7
    
8.
Singh V, John KM, Malik A, Pareek T, Dutta V. Xanthogranulomatous appendicitis: Uncommon histological variant of a common entity. Med J Armed Forces India 2015;71 Suppl 1:S19-21.  Back to cited text no. 8
    
9.
Guo G, Greenson JK. Histopathology of interval (delayed) appendectomy specimens: Strong association with granulomatous and xanthogranulomatous appendicitis. Am J Surg Pathol 2003;27:1147-51.  Back to cited text no. 9
    
10.
Kochhar G, Saha S, Andley M, Kumar A, Kumar A. Xanthogranulomatous appendicitis with a fulminant course: Report of a case. J Clin Diagn Res 2014;8:ND01-2.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

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