|Year : 2014 | Volume
| Issue : 2 | Page : 228-230
Monika Rathi1, Jyotsana Khattri2, Satish Kumar Budania3, Jaskirat Singh2, Seema Awasthi2, Sushmit Verma4
1 Department of Pathology, Lala Lajpat Rai Memorial Medical College, Meerut; Teerthanker Mahaveer Medical College and Research Center, Moradabad, Uttar Pradesh, India
2 Teerthanker Mahaveer Medical College and Research Center, Moradabad, Uttar Pradesh, India
3 Department of Psychiatry, Subharti Medical College, Meerut, Uttar Pradesh, India
4 Department of Orthopaedics, Lala Lajpat Rai Memorial Medical College, Meerut, Uttar Pradesh, India
|Date of Web Publication||11-Nov-2014|
Teerthanker Mahaveer Medical College and Research Center, 228/16, Saket Colony, North Civil Lines, Muzaffarnagar, Moradabad - 251 001, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
Xanthogranulomatous inflammation in bone is a very rare entity. We came across this rare case of xanthogranulomatous osteomyelitis. We present a rare case of 50-year-old male who presented to the hospital with the chief complaint of pain and swelling in left ankle for 5 months and fever for 2 days. Total leukocyte count and differential leucocyte count was normal. Renal function tests and liver function tests were normal. Montoux test was negative. Chest X-ray was normal. Pus culture was negative for acid-fast bacilli but showed heavy growth of Pseudomonas. X-ray imaging revealed soft tissue mass, periosteal reaction and cortical disruption in the lower end of left tibia. Sensitivity to Amikacin and Imipenem was found on pus culture. So patient was started with these antibiotics. However, pain was not relieved. So arthrodesis was done, and curetted material was sent to histopathology lab where it was diagnosed xanthogranulomatous osteomyelitis. The patient was followed-up for 1 month. He did not have any complaints. All the previously existing complaints subsided. Xanthogranulomatous inflammation can be seen in bones also, though rarely reported.
Keywords: Arthodesis, xanthogranulomatous osteomyelitis, tibia
|How to cite this article:|
Rathi M, Khattri J, Budania SK, Singh J, Awasthi S, Verma S. Xanthogranulomatous osteomyelitis
. Arch Med Health Sci 2014;2:228-30
| Introduction|| |
Xanthogranulomatous inflammation has been described in various organs such as kidney, gallbladder, colon, pancreas, and salivary gland. 
However, xanthogranulomatous osteomyelitis has not been much described in the literature. Organs such as lung, brain, and bone are rarely affected. Bone involvement is accompanied by systemic and regional clinical presentations such as pain, fever, and leukocytosis.  Hence far seven cases of xanthogranulomatous osteomyelitis have been reported in the world to the best of our knowledge. ,,,,, We report the eighth case of xanthogranulomatous osteomyelitis.
| Case Report|| |
The patient provided consent to report this case.
A 50-year-old male was admitted to the hospital with the chief complaint of pain and swelling in left ankle for 5 months and fever for 2 days. Patient was diagnosed to be suffering from tuberculosis 4 years back for which he had completed full course of antituberculous treatment (isoniazid, rifampicin, ethambutol, and pyrazinamide daily for 2 months, followed by 4 months of isoniazid and rifampicin).
Physical examination showed tenderness and swelling in the ankle with a pus point on swelling.
Laboratory investigation showed raised erythrocyte sedimentation rate. Total leucocyte count and differential leukocyte count was normal. Renal function tests and liver function tests were normal. Montoux test was negative. Chest X-ray was negative.
X-ray imaging revealed soft tissue mass, periosteal reaction and cortical disruption in the lower end of left tibia [Figure 1].
On microbiological examination, pus culture was negative for acid fast bacilli but showed heavy growth of Pseudomonas. Sensitivity to Amikacin and Imipenem was found. Hence, the patient was started on these antibiotics. However, the pain was not relieved, and arthodesis was done. Curated material was sent to histopathology lab.
Grossly, the specimen received was soft and gray-brown. Microscopically, mixed inflammatory infiltrate comprised of sheets of foamy histiocytes, lymphocytes, plasma cells, and few polymorphs was seen infiltrating the bony trabeculae. Stain for acid-fast bacilli was negative. Thus, a histopathological diagnosis of xanthogranulomatous osteomyelitis was made [Figure 2]a-d.
|Figure 2: (a) Bony trabeculae (H and E, ×100). (b) Chronic inflammatory infiltrate (H and E, ×400). (c) Xanthogranulomatous change (H and E, ×100). (d) Xanthogranulomatous change (H and E, ×400)|
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| Discussion|| |
Thus, xanthogranulomatous osteomyelitis is a rare entity. Delayed-type hypersensitivity reaction of cell-mediated immunity may be implicated in its pathogenesis. Grossly, it is a mass-like lesion extending to enclosing tissues, which can mimic the infiltrative cancer. 
Microscopically, it shows the presence of features of osteomyelitis (admixture of inflammatory cells including neutrophils, lymphocytes, plasma cells) with abundant macrophages. 
Although association between bacterial infection and the xanthogranulomatous inflammation has been determined in several organs such as kidneys, but it is still to be proven for bone. 
The first two cases of xanthogranulomatous osteomyelitis were diagnosed by Cozzutto in 5-year and 14-year-old males in fist rib and tibia respectively.  Borjian et al. described a case of xanthogranulomatous osteomyelitis involving two bones independently, one in the right humerus and other in left fibula, simultaneously. 
The key features of previously diagnosed cases described in the literature are discussed in [Table 1].
|Table 1: Salient features of reported cases of xanthogranulomatous osteomyelitis|
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Thus, majority of the patients presented with fibrile illness, pain, and swelling in the bones.
Laboratory investigations revealed the neutrophilic leucocytosis and culturable micro-organism in few patients. X-ray revealed osteolytic lesions in most patients.
Microscopically, xanthogranulomatous osteomyelitis has to be differentiated from Langerhan's cell histiocytosis, Erdheim-Chester disease, and lipid storage disorders. Langerhan's cell histiocytosis commonly affects mid-shaft portions of long bones and often has infiltration of eosinophils. Erdheim-Chester disease is a multifocal disorder with frequent involvement of extraskeletal tissues. Histology shows presence of cholesterol clefts along with foamy histiocytes. In the case of Gaucher's disease and Niemann Pick disease, foam cell transformation is usually seen within the bone marrow, and a clinical history plays an important role in differentiating storage disorders from xanthogranulomatous osteomyelitis. 
Thus, we conclude that xanthogranulomatous osteomyelitis is a rare diagnosis which can be treated easily. Hence, care has to take to differentiate it from other benign and malignant lesions of bone.
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[Figure 1], [Figure 2]