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Table of Contents
CASE REPORT
Year : 2020  |  Volume : 17  |  Issue : 4  |  Page : 275-276

Xanthogranulomatous osteomyelitis


1 Department of Pathology, Indraprastha Apollo Hospital, New Delhi, India
2 Department of Orthopedics, Indraprastha Apollo Hospital, New Delhi, India

Date of Submission19-Nov-2019
Date of Acceptance30-Dec-2019
Date of Web Publication21-Dec-2020

Correspondence Address:
Seema Singhal
Department of Pathology, Indraprastha Apollo Hospital, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/am.am_72_19

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  Abstract 


Xanthogranulomatous osteomyelitis (XO) is a type of chronic osteomyelitis characterized by collection of foamy macrophages admixed with mononuclear cells. Xanthogranulomatous inflammation is characterized by the presence of histiocytes, foamy macrophages, activated plasma cells, and presence of suppurative foci and hemorrhage. It has been encountered in various tissues such as gallbladder, kidney, urinary bladder, fallopian tube, ovary, vagina, prostate, testis, epididymis, colon, and appendix. Very rarely, it can affect lungs, brain, or bone. Only three cases of XO have been described previously in the literature till date.

Keywords: Histiocytic, osteomyelitis, xanthogranulomatou


How to cite this article:
Singhal S, Shekher C. Xanthogranulomatous osteomyelitis. Apollo Med 2020;17:275-6

How to cite this URL:
Singhal S, Shekher C. Xanthogranulomatous osteomyelitis. Apollo Med [serial online] 2020 [cited 2021 Jan 18];17:275-6. Available from: https://www.apollomedicine.org/text.asp?2020/17/4/275/304272




  Introduction Top


Xanthogranulomatous inflammation is characterized by the presence of histiocytes, foamy macrophages, activated plasma cells and presence of suppurative foci and hemorrhage.


  Case Report Top


A 26-year-old male presented with complaints of pain and swelling middle of the left arm for 2 months.

On local examination, there was a small nontender prominence of 3.5 cm × 3 cm located in the upper and middle part of the left humerus.

Anteroposterior X-ray of the left humerus revealed the presence of bony sequestrum [Figure 1]a. Surrounding soft tissues were normal. Radiological findings were suggestive of osteomyelitis. Systemic examination was normal.
Figure 1: (a) X-ray (anteroposterior view left humerus): Bony sequestrum, rarefaction of humerus shaft. (b) Sheets of foamy histiocytes, macrophages with interspersed foci of collection of neutrophils, hemorrhage, and plasma cells (H and E, ×200)

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Alkaline phosphatase was elevated to levels of 400 IU/L (normal: 100–280 IU/L), and liver and renal function tests were within normal limits.

Core needle biopsy was done to ascertain the disease process. Tissue sections showed the presence of sheets of foamy histiocytes, macrophages with interspersed foci of collection of neutrophils, hemorrhage, and plasma cells [Figure 1]b.


  Discussion Top


Chronic osteomyelitis develops in 15%–30% of cases. It occurs due to lack of treatment, inadequate antibiotic treatment, or incomplete surgical debridement of necrotic bone. Xanthogranulomatous osteomyelitis (XO) is a type of chronic osteomyelitis and may mimic malignancy radiologically. Xanthogranulomatous osteomyelitis (XO) is a type of chronic osteomyelitis characterized by collection of foamy macrophages admixed with mononuclear cells.[1]

Xanthogranulomatous inflammation has been described in various tissues such as gallbladder, kidney, urinary bladder, fallopian tube, ovary, vagina, testis, epididymis, colon, and appendix.[4],[5],[6] Rarely, it is also seen in the lungs, brain, and prostate. It is a great mimicker of malignancy on gross and radiological examination. Exact pathogenesis is unknown; however, defective lipid transport, immunological disorders, low-virulent infections, reactions to specific infectious agents, and lymphatic obstruction have been proposed as putative mechanisms.[7] McVey and McMahon suggested that xanthogranulomatous inflammation is related to a long-standing or recurrent inflammatory process.[8] With respect to the origin of foam cells, it is considered that the vast majority of foam cells are derived from monocytes/macrophages. Delayed-type hypersensitivity reaction of cell-mediated immunity may be implicated in the pathogenesis of XO.

This term was coined due to the similar histopathological picture in bone as seen in other organs.[2]

Tissue pathology reveals the chronic inflammatory nature of the disease. Only three cases have been described till date in the medical literature.[2],[3] The two cases described by Cozzutto were in patients in the first two decades of life and involved the first rib and the tibial epiphysis, respectively,[2] whereas the third case described by Vankalakunti et al.[3] was in a 50-year-old woman with involvement of ulna.

Our case was seen in a 26-year male with involvement of humerus.

The differential diagnoses include chronic recurrent multifocal osteomyelitis, xanthoma, infiltrative storage disorder, malakoplakia, Langerhans cell histiocytosis, fibrohistiocytic tumor, Erdheim–Chester disease, and metastatic renal cell carcinoma.[9],[10],[11],[12]

In this case, the presence of sheets of foamy macrophages, macrophages with interspersed foci of collection of neutrophils, hemorrhage, and plasma cells on tissue sections confirmed the diagnosis.


  Conclusion Top


This case is primarily presented due to its rarity. XO has been sparingly reported in the literature. XO may simulate a tumor, either primary or secondary, both on radiology and gross examination. A bone biopsy as done in this case might prevent unnecessary surgery/amputation and hence imparting appropriate treatment.

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.
Cozzutto C, Carbone A. The xanthogranulomatous process. Xanthogranulomatous inflammation. Pathol Res Pract 1988;183:395-402.  Back to cited text no. 1
    
2.
Cozzutto C. Xanthogranulomatous osteomyelitis. Arch Pathol Lab Med 1984;108:973-6.  Back to cited text no. 2
    
3.
Vankalakunti M, Saikia UN, Mathew M, Kang M. Xanthogranulomatous osteomyelitis of ulna mimicking neoplasm. World J Surg Oncol 2007;5:46.  Back to cited text no. 3
    
4.
Ladefoged C, Lorentzen M. Xanthogranulomatous cholecystitis. A clinicopathological study of 20 cases and review of the literature. APMIS 1993;101:869-75.  Back to cited text no. 4
    
5.
Nistal M, Gonzalez-Peramato P, Serrano A, Regadera J. Xanthogranulomatous funiculitis and orchiepididymitis: Report of 2 cases with immunohistochemical study and literature review. Arch Pathol Lab Med 2004;128:911-4.  Back to cited text no. 5
    
6.
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. 6
    
7.
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. 7
    
8.
McVey RJ, McMahon RF. Xanthogranulomatous appendicitis. Histopathology 1994;24:198.  Back to cited text no. 8
    
9.
Hamada T, Ito H, Araki Y, Fujii K, Inoue M, Ishida O. Benign fibrous histiocytoma of the femur: Review of three cases. Skeletal Radiol 1996;25:25-9.  Back to cited text no. 9
    
10.
Kossard S, Chow E, Wilkinson B, Killingsworth M. Lipid and giant cell poor necrobiotic xanthogranuloma. J Cutan Pathol 2000;27:374-8.  Back to cited text no. 10
    
11.
Girschick HJ, Huppertz HI, Harmsen D, Krauspe R, Müller-Hermelink HK, Papadopoulos T. Chronic recurrent multifocal osteomyelitis in children: Diagnostic value of histopathology and microbial testing. Hum Pathol 1999;30:59-65.  Back to cited text no. 11
    
12.
Kayser R, Mahlfeld K, Grasshoff H. Vertebral Langerhans-cell histiocytosis in childhood: A differential diagnosis of spinal osteomyelitis. Klin Padiatr 1999;211:399-402.  Back to cited text no. 12
    


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