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| CASE REPORT |
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| Year : 2018 | Volume
: 6
| Issue : 1 | Page : 120-121 |
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Xanthogranulomatous appendicitis: Entity of surprise
Anindya Adhikari, Rudra Narayan Ray, Raison Shail Minz, Bidhan Chandra Nayek
Department of Pathology, Bankura Sammilani Medical College, Bankura, West Bengal, India
| Date of Web Publication | 11-Jun-2018 |
Correspondence Address:
Dr. Anindya Adhikari
Vil-Basudevpur, P.O-Banipur, P.S-Sankrail, Howrah - 711 304, West Bengal
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/amhs.amhs_43_18
Xanthogranulomatous inflammation is a special type of inflammatory process with unknown etiology. Commonly involved organs are gallbladder and kidney. So far, only a negligible number of cases of xanthogranulomatous appendicitis (XA) have been reported in different literatures. Herein, we report a case of XA in a 49-year-old female patient, who presented with recurrent right iliac fossa pain. Clinically, appendicitis was diagnosed and appendectomy was performed. Histopathological examination revealed it as a case of XA. No other form of treatment was given and the patient is doing well 1 month postoperatively.
Keywords: Appendicitis, inflammation, xanthogranulomatous
How to cite this article:
Adhikari A, Ray RN, Minz RS, Nayek BC. Xanthogranulomatous appendicitis: Entity of surprise. Arch Med Health Sci 2018;6:120-1 |
| Introduction | |  |
Xanthogranulomatous process (XP), also known as xanthogranulomatous inflammation, is a form of acute and chronic inflammation characterized by well-recognized clustering of foamy macrophages among other inflammatory cells. This entity was first described in kidney by Osterlind in 1944.[1] Although various other organs can be affected, the occurrence of xanthogranulomatous appendicitis (XA) is unusual.
| Case Report | | |
A 49-year-old female presented with complaints of recurrent right iliac fossa pain for the last 6 months. The pain was insidious in onset with subtle progression. It was a localized pain, throbbing in character each time. In the last episode of attack of pain, she had mild fever, vomiting, and mild burning urinary sensations also, which persisted for 2 days. She had no addiction and had no significant past medical or surgical history. Family history was unremarkable too. On general survey, the patient had average build and nutrition. On physical examination, she had a temperature of 100°F, pulse rate of 90/min, mild dehydration, and tender McBurney's point. Routine blood test revealed slightly high total leukocyte count (12,200/dl). Ultrasonography (USG) of abdomen performed 4 months back during the second attack was unremarkable. This time USG was not arranged as the patient came at midnight. On clinical ground, diagnosis of acute on chronic appendicitis was rendered and appendectomy was performed without delay. Grossly, the appendix was found wrapped intimately with omentum all around. Whole specimen [Figure 1]a was measuring 7 cm × 5 cm. Appendix was small - 2.5 cm in length and 0.5 cm in width. Its surface was congested. Omental tissue was firm nodular in feel. A cut section demonstrated no fecolith. Histopathological examination on hematoxylin–eosin stain showed discontinuous eroded appendicular mucosa. Submucosa including muscularis propria revealed plenty of foamy macrophages and multinucleated foreign body type of giant cells [Figure 1]b. Chronic inflammatory cells (lymphocytes and plasma cells) and eosinophils were also noted. There was focal aggregate of cholesterol crystals [Figure 2]a and occasional lymphoid follicle [Figure 2]b. Omental tissue on histopathological examination showed congested blood vessels only. Based on morphology, a diagnosis of XA was rendered. No postoperative therapy was given. One-month follow-up period was uneventful. | Figure 1: (a) Gross appearance of the specimen showing small appendix wrapped by the omentum. (b) Giant cell and aggregate of foamy macrophages
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 | Figure 2: (a) Aggregate of cholesterol crystals. (b) Lymphoid follicle and foamy macrophages
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| Discussion | | |
XP is a well-defined form of acute and chronic inflammation. In 1993, Birch et al. published the first reported cases of XA.[2] It is manifested by the presence of lipid-laden macrophages admixed with lymphocytes, plasma cells, neutrophils, and often multinucleated giant cells with or without cholesterol clefts.[3] Kidney and gallbladder are the organs most commonly involved.[4] Some cases of XP have been reported in other organs such as endometrium, epididymis, Fallopian tube More Detailss, bone, skin, appendix, urinary bladder, and thyroid and adrenal glands.[5] XA is a rare phenomenon and it occurs in adults with a mean age of 47.9 years (83%, 21–78 years).[6] The exact etiology of XP is unknown. Proposed theories include defective lipid transport, immunologic disturbances, infection by low-virulence organisms, and lymphatic obstruction.[7] Cozzutto and Carbone noted that XP is usually seen secondary to inflammation, hemorrhage, and necrosis.[3] A study of appendectomies for a period of 4 years by Guo and Greenson found XA in 8 (36.4%) of 22 cases of interval appendicectomies, whereas no such cases were seen in emergency appendicectomies.[6] XA is a rare occurrence in resected specimens of acute appendicitis.[4] Munichor et al. reported a case of XA in a 37-year-old female, who presented with typical signs and symptoms of acute appendicitis.[7] Singh et al. also reported XA in a 21-year-old woman, who presented clinically with acute appendicitis.[8] Our case had acute on chronic presentation. XA should be differentiated from other lesions containing foam cells and granulomatous inflammation such as malakoplakia, localized xanthoma, and Crohn's disease. In the localized xanthoma, there is only aggregate of foam cells without any parenchymal destruction and inflammatory reaction.[8] The presence of inflammation along with the absence of transmural involvement by granulomas and Michaelis–Gutmann bodies ruled out localized xanthoma, Crohn' s disease, and malakoplakia, respectively, in our case.
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
The study was supported by the Department of Pathology, Bankura Sammilani Medical College, Bankura, West Bengal, India.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Osterlind S. Uber pyelonephritis xanthomatosa. Acta Chir Scand 1994;90:369-76.  |
| 2. | Birch PJ, Richmond I, Bennett MK. Xanthogranulomatous appendicitis. Histopathology 1993;22:597-8. [ PUBMED] |
| 3. | Cozzutto C, Carbone A. The xanthogranulomatous process. Xanthogranulomatous inflammation. Pathol Res Pract 1988;183:395-402. [ PUBMED] |
| 4. | Mehrotra N, Arakeri SU. Xanthogranulomatous appendicitis: A rare case report. JKIMSU 2017;6:125-8. |
| 5. | 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. |
| 6. | Thapa R, Gurung P, Kafle N, Pradhanang S, Lakhey M, Singh DR. Xanthogranulomatous appendicitis: A case report. J Inst Med 2016;38:2-3. |
| 7. | Munichor M, Kerner H, Cohen H, Bickel A, Iancu TC. Xanthogranulomatous appendicitis – An incidental finding of localized pathology. Ultrastruct Pathol 2000;24:33-9. [ PUBMED] |
| 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:S19-21. [ PUBMED] |
[Figure 1], [Figure 2]
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