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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 9  |  Issue : 4  |  Page : 177-179

Xanthogranulomatous appendicitis: A rare case


Department of General Surgery, Mallareddy Institute of Medical Sciences, Hyderabad, Telangana, India

Date of Submission27-Mar-2021
Date of Decision09-Jun-2021
Date of Acceptance24-Jun-2021
Date of Web Publication26-Nov-2021

Correspondence Address:
Dr. Koilakonda Ajay Kumar
Department of General Surgery, Mallareddy Institute of Medical Sciences, Hyderabad, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjhs.mjhs_24_21

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  Abstract 


Xanthogranulomatous appendicitis (XA) is a rare type of presentation of acute appendicitis. Only few cases of XA have been reported in the literature so far. Occurrence of xanthogranulomatous inflammation is rare in the appendix even though it is more common in the gallbladder and kidney. We report a case of 35-year-old male who presented with typical signs of acute appendicitis and needed an immediate laparotomy. Histopathological examination revealed the diagnosis of XA. Although the condition is rare, because of curability unlike the malignancy which requires extensive surgery, it should be considered in differential diagnosis while encountered with appendicular mass lesions.

Keywords: Acute appendicitis, case report, xanthogranulomatous appendicitis, xanthogranulomatous inflammation


How to cite this article:
Kumar KA, Kavali H, Iswarya J, Prasad P K. Xanthogranulomatous appendicitis: A rare case. MRIMS J Health Sci 2021;9:177-9

How to cite this URL:
Kumar KA, Kavali H, Iswarya J, Prasad P K. Xanthogranulomatous appendicitis: A rare case. MRIMS J Health Sci [serial online] 2021 [cited 2022 Aug 18];9:177-9. Available from: http://www.mrimsjournal.com/text.asp?2021/9/4/177/331238




  Introduction Top


Xanthogranulomatous inflammation (XGI) is a rare form of chronic inflammation characterized histologically by the presence of high number of foamy histiocytes admixed with lymphocytes and plasma cells, first reported in the genitourinary tract and gallbladder.[1] It can involve any organ, but the most common sites are kidney and gallbladder. Although acute appendicitis is a very common surgical condition, xanthogranulomatous appendicitis (XA) is a rare phenomenon. Only few cases have been reported so far in the literature in which XGI involved the appendix and the patient presented as a case of acute appendicitis. XA may mimic locally advanced cancer but has a benign course and can be cured by surgical resection.


  Case Report Top


A 35-year-old male with no known comorbidities presented with complaint of pain abdomen for the past 3 days, which was insidious in onset associated with two episodes of vomiting for the past 1 day and fever for the past 1 day. On clinical examination, the patient had McBurney point tenderness, Rovsing's sign positive, and rebound tenderness positive. With clinical suspicion of acute appendicitis, Ultrasonography (USG) of abdomen done showed signs suggestive of acute appendicitis with appendicular mass formation.

Diagnosis of acute appendicitis was rendered, and the patient was taken for emergency open appendicectomy with consent. Intraoperatively, inflamed appendix with tip of the appendix enlarged, and appendix was adhered to posterior abdominal wall and partly to terminal ileum. Appendix was stony hard in consistency and resembled appendicular tumor. Appendicular base being normal, appendicectomy was done and hemostasis secured.

Whole specimen of the appendix of length 5.5 cm with enlarged and expanded tip of about 3.5 cm × 3 cm with patent lumen was sent for histopathological examination (HPE) [Figure 1]. HPE of multiple sections revealed appendix with ulceration of the mucosa at places and histological morphology suggestive of XGI [Figure 2].
Figure 1: Excised appendix specimen

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Figure 2: Histopathological examination showing xanthogranulomatous changes

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Diagnosis of XA was rendered. No postoperative therapy was given. Six months of follow-up period was uneventful.


  Discussion Top


XGI is a pathologic entity with unique and characteristic macroscopic and microscopic features. Typical findings include bright yellow or golden yellow mass-like lesions on macroscopic examination associated with abscess cavities, microabscesses, and large numbers of lipid-laden macrophages; as well a minor component of chronic and acute inflammatory cells on microscopic examination; mimicking locally advanced carcinoma.

XGI of the appendix is rare. In 1993, Birch et al. published the first reported cases of XA.[2] Microscopic examination of XGI 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. The cholesterol clefts were not seen, but reactive lymphoid hyperplasia was seen in our case. XGI causes destruction and effacement of the normal structures of the involved organ and could be misinterpreted as a locally invasive cancerous lesion.

Involvement of the appendix by XGI, or XA, is a rare phenomenon with only few cases reported in the literature. Most of these case reports were described in adults (mean age = 42.3 years, range is from 12 to 78 years). One study described the case of a 12-year-old boy who had XA in an interval appendectomy, and the specimen was removed 6 weeks after an episode of acute appendicitis.[2] In most of the cases, XGI was seen in interval appendicectomy specimens.[3],[4]

The aim of this case presentation is to report one of the rare presentations of acute appendicitis, i.e. XA.


  Conclusion Top


XA is a rare benign condition causing signs and symptoms of acute appendicitis. XA mimics as a locally advanced malignant lesion but has a benign course, which can be managed by surgical resection of lesion unlike malignancy which may require right hemicolectomy. Although the condition is rare because of curability unlike the malignancy which requires extensive surgery, it has to be kept in mind as one of the differentials while encountered with appendicular mass lesions.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Franco V, Aragona F, Genova G, Florena AM, Stella M, Campesi G. Xanthogranulomatous cholecystitis. Histopathological study and classification. Pathol Res Pract 1990;186:383-90.  Back to cited text no. 1
    
2.
Birch PJ, Richmond I, Bennett MK. Xanthogranulomatous appendicitis. Histopathology 1993;22:597-8.  Back to cited text no. 2
    
3.
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:S19-21.  Back to cited text no. 3
    
4.
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. 4
    


    Figures

  [Figure 1], [Figure 2]



 

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