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  Table of Contents    
LETTER TO EDITOR  
Year : 2016  |  Volume : 59  |  Issue : 2  |  Page : 253-254
Xanthogranulomatous osteomyelitis mimicking neoplasm: A rare entity


1 Department of Pathology, Apollo Hospitals, Bhubaneswar, Odisha, India
2 Department of Radiology, Apollo Hospitals, Bhubaneswar, Odisha, India

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Date of Web Publication9-May-2016
 

How to cite this article:
Baisakh MR, Kar MR, Agrawal A, Mohapatra N. Xanthogranulomatous osteomyelitis mimicking neoplasm: A rare entity. Indian J Pathol Microbiol 2016;59:253-4

How to cite this URL:
Baisakh MR, Kar MR, Agrawal A, Mohapatra N. Xanthogranulomatous osteomyelitis mimicking neoplasm: A rare entity. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Dec 10];59:253-4. Available from: http://www.ijpmonline.org/text.asp?2016/59/2/253/182016


Editor,

Xanthogranulomatous osteomyelitis (XO) is a rare form of chronic inflammatory disorder clinically characterized by pain, fever, and leukocytosis and often mimics neoplastic lesions. A 21-year-old female presented with pain in bilateral lower limb toward the lower thigh and around the knee since last 1 year associated with on and off fever. On examination, there was tenderness over bone more on the right side than the left. Bilateral X-ray showed lytic lesion in the upper end of the tibia and the lower end of the femur. CT scan report showed large expansile lytic lesion involving the lower shaft of the femur and the upper shaft of the tibia on both sides (involving diaphysis, metaphysis, and epiphyseal regions) with range of differential diagnoses, i.e., metastasis, eosinophilic granuloma, multicentric chronic infections, and hyperparathyroidism [Figure 1]. MRI scan revealed bilateral hyperintense irregularly outlined lesion involving dia-meta-epiphyseal region of tibia and femur on T1-weighted image suggestive of metastasis. Routine hematological investigations showed mild anemia (Hb - 9.4 g/dl), mild leukocytosis (16,200/cumm), increased erythrocyte sedimentation rate (70 mm in 1st h), and increased C-reactive protein. Biochemical investigation showed normal serum PTH and calcium level. Bone marrow aspiration revealed 8% plasmacytosis. Serum electrophoresis revealed hypoalbuminemia with increase beta and gamma globulinemia. The patient was advised for image-guided core needle biopsy from the lytic lesion in the femur. Histopathology showed sheets of foamy macrophages closely intermingled with plasma cells, lymphocytes, and occasional polymorphs. Focal irregular fragments of dead bones were noted [Figure 2]. No atypia was noted. Periodic acid-Schiff and Mucicarmine stains done were negative. Correlating clinical, radiological, and serological findings a diagnosis of XO was rendered. Culture and sensitivity revealed no organism.
Figure 1: (a) Bilateral lytic lesion involving both tibia and femur in T1-weighted image of magnetic resonance imaging. (b) T2-weighted image showing marked epi-meta-diaphyseal lesion. (c) Computed tomography scan showing expansile lytic lesion with irregular border involving femur. (d) Tibia showing focal cortical break

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Figure 2: (a) Sheets of foamy macrophages closely admixed with plasma cells and lymphocytes. (b) Focal fragment of irregular bony tissue

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Xanthogranulomatous lesion is a chronic inflammatory process characterized histologically by abundant foamy histiocytes, multinucleated giant cells, and fibrosis together with plasma cells, lymphocytes, and polymorphonuclear cells. This lesion has been described in different organs such as gallbladder, kidney, pancreas, fallopian tube, ovary, epididymis, testis, prostate, and salivary gland.[1],[2] It occurs very rarely in brain, lung, and bone.[2]

To the best of our knowledge, only 8 cases of XO have been described worldwide. Cozzutto described 2 cases of XO for the first time in 1984.[3] Multifocal bone involvements such as our case have been described by only Borjian et al. and Cennimo et al.[4],[5]

Its pathogenesis is attributed to delayed type hypersensitivity reaction of cell-mediated immunity. Though bacterial infection found to be associated with xanthogranulomatous lesions of many organ sites, its role in bone is yet to be established.[3]

XO produces mass-like lesion grossly and radiologically, hence often mimics infiltrative malignant tumor. Therefore, differentiating this pseudo-neoplasm of infective etiology from tumor is an important step in diagnosis. The list differential diagnosis includes Langerhans cell histiocytoses, Erdheim–Chester disease, chronic recurrent multifocal osteomyelitis, xanthoma, infiltrative storage disorder, malakoplakia, fibrohistiocytic tumor, and metastatic renal cell carcinoma. Clinical presentation, radiological picture, and histopathological findings together are important in diagnosis of XO. However, histopathology is gold standard in final diagnosis of the disease.

We presented this case because of its rarity. Awareness of such entity and correct diagnosis in histopathology are crucial in appropriate management of this disease.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
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. 1
    
2.
Rafique M, Yaqoob N. Xanthogranulomatous prostatitis: A mimic of carcinoma of prostate. World J Surg Oncol 2006;4:30.  Back to cited text no. 2
    
3.
Cozzutto C. Xanthogranulomatous osteomyelitis. Arch Pathol Lab Med 1984;108:973-6.  Back to cited text no. 3
    
4.
Borjian A, Rezaei F, Eshaghi MA, Shemshaki H. Xanthogranulomatous osteomyelitis. J Orthop Traumatol 2012;13:217-20.  Back to cited text no. 4
    
5.
Cennimo DJ, Agag R, Fleegler E, Lardizabal A, Klein KM, Wenokor C, et al. Mycobacterium marinum hand infection in a “Sushi Chef”. Eplasty 2009;9:e43.  Back to cited text no. 5
    

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Correspondence Address:
Manas R Baisakh
pollo Hospitals, 251, Sainik School Road, Unit-15, Bhubaneswar - 751 005, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.182016

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