Multifocal bilateral xanthogranulomatous osteomyelitis

Sapra, Rahul; Jain, Pankaj; Gupta, Shubha; Kumar, Rakesh

doi:10.4103/0019-5413.159682

Cited by 8 publications

(4 citation statements)

References 7 publications

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“…Xanthogranulomatous inflammation can extend into the adjacent surrounding structure; therefore, there are a lot of reports of XGI directly infiltrating multiple close organs. Although there have been a few reported cases of XGI confirmed on multiple sites such as multifocal xanthogranulomatous osteomyelitis 25 or bilateral xanthogranulomatous orchiepididymitis, 26 xanthogranulomatous change independently confirmed on the multiple organs as our case is extremely rare. Although XGP is a benign condition, almost all reported cases underwent surgical treatment based on the presurgical diagnosis of malignant tumors (13 of 31), pseudocysts (5 of 31), IPMNs (5 of 31), pancreatitis-related nonspecific inflammatory lesions (4 of 31), and SPNs (2 of 31).…”

Section: Discussion and Literature Reviewmentioning

confidence: 62%

Xanthogranulomatous Pancreatitis Accompanied by Xanthogranulomatous Cholecystitis

et al. 2021

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Xanthogranulomatous pancreatitis (XGP) is extremely rare, with only 31 cases reported in the English literature to date. We reviewed previously reported 17 articles about XGP and report an additional case of XGP. This is the first case of XGP with xanthogranulomatous cholecystitis accompanied by intraductal papillary mucinous carcinoma (IPMC) in an 80-year-old woman. She was referred to our hospital with jaundice and general malaise and was found to have a cystic mass at the pancreatic head and a solid mass at the pancreatic tail, with dilation of both the main pancreatic duct and biliary tract noted on computed tomography. Diagnosis of IPMC at the pancreatic head with neuroendocrine tumor at the pancreatic tail was made, and the patient underwent subtotal stomach-preserving pancreatoduodenectomy with enucleation of the mass at the tail. Pathological examination revealed IPMC with xanthogranulomatous changes around the IPMC and at the pancreatic tail and gallbladder. Xanthogranulomatous pancreatitis could be induced by inflammatory reaction due to obstruction of the pancreatic duct and biliary tract by mucin produced in the IPMC. It is sometimes difficult to preoperatively differentiate XGP from malignant pancreatic tumors, such as pancreatic carcinoma or neuroendocrine tumor, using imaging studies.

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Section: Discussion and Literature Reviewmentioning

confidence: 62%

Xanthogranulomatous Pancreatitis Accompanied by Xanthogranulomatous Cholecystitis

et al. 2021

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“…XO was first introduced in the reports of 2 cases in 1984 followed by reports of 18 other patients summarized in the Table. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] A review of these 20 cases and the case of our patient suggests that XO is more prevalent in males (15 of 21) than females and has no characteristic age distribution. Most patients (17 of 21) had unifocal XO; only 4 had multifocal lesions.…”

Section: Discussionmentioning

confidence: 99%

“…Most patients (17 of 21) had unifocal XO; only 4 had multifocal lesions. 7,10,17,20 The most commonly affected sites are the long tubular bones (17 of 21). As with our patient, pain and swelling of the involved bone seem to be the primary reason for presentation.…”

Section: Discussionmentioning

confidence: 99%

“…As with our patient, pain and swelling of the involved bone seem to be the primary reason for presentation. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] Cheema et al attributed the pathogenesis of xanthogranulomatous process in bone to a delayed-type hypersensitivity reaction of cell-mediated immunity. 22 Trauma has been also mentioned as a hypothetical reason for this process.…”

Section: Discussionmentioning

confidence: 99%

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Xanthogranulomatous Osteomyelitis of the Tibia

Solooki

Hoveidaei

Kardeh

et al. 2019

TOJ

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Background: Xanthogranulomatous osteomyelitis (XO) is a rare chronic inflammatory process that is histologically characterized by the presence of foamy macrophages, histiocytes, and plasma cells. Radiologic and gross examinations can mimic malignancy, so definitive diagnosis should be made by histopathologic evaluation. Case Report: A 15-year-old male presented with pain in the proximal right leg for 2 weeks prior to admission. The patient had a history of leg trauma 3 years prior that was responsive to as-needed analgesics. Laboratory data revealed increased erythrocyte sedimentation rate and C-reactive protein. X-ray of the right tibia showed a periosteal reaction and bulging of bone with a questionable destruction of the cortex. Magnetic resonance imaging demonstrated an expansile bony lesion in the proximal metaphysis of the tibia. Histopathologic examination showed the bone trabecula surrounded by chronic inflammatory cells and a fragment of dead bone surrounded by histiocytes, foamy macrophages, and plasma cells. The pathologist confirmed the diagnosis of XO. Wound culture proved moderate growth of Staphylococcus aureus, and treatment with cefazolin and cephalexin was successful. Conclusion: The patient was not treated with the routine therapy used in previously reported cases (curettage), suggesting that antibiotic therapy should be considered before employing surgical interventions for XO.

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Xanthogranulomatous osteomyelitis of the humerus in a pediatric patient with Alagille syndrome: a case report and literature review

2017

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Xanthogranulomatous osteomyelitis (XO) is an exceedingly rare disease characterized by infiltration of histiocytes and foamy macrophages. Both on gross examination and on radiographs, XO can mimic malignancy. We describe the case of a 5-year-old female with Alagille syndrome who presented with a pathologic fracture of the right humerus. Initial radiographs revealed multiple osteolytic lesions in the distal humerus while MRI showed a large soft tissue mass. Biopsy confirmed the diagnosis of XO, which has hitherto not been described in a patient with Alagille syndrome. The patient was admitted for IV antibiotics and taken to the operating room for an incision and debridement. Tissue cultures were obtained and grew Salmonella. Antibiotic therapy was tapered, and the patient responded to treatment. She was doing well at her 6-month follow-up visit. In the discussion section, we explore how osteopenia and immune dysregulation caused by Alagille syndrome can affect the development of XO. We summarize all previously reported cases of XO and conclude that XO presents as an osteolytic lesion that expands rapidly over the course of a few weeks. We highlight that XO can mimic sarcoma because of its mass effect but can be distinguished radiographically by the presence of surrounding sclerosis. Given the rapid onset of XO, we classify it as an acute osteomyelitis. We discuss how leukemia and Ewing sarcoma can present similarly to acute osteomyelitis. We then emphasize key features that can be used to distinguish these malignancies from acute osteomyelitis.

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Multifocal bilateral xanthogranulomatous osteomyelitis

Cited by 8 publications

References 7 publications

Xanthogranulomatous Pancreatitis Accompanied by Xanthogranulomatous Cholecystitis

Xanthogranulomatous Pancreatitis Accompanied by Xanthogranulomatous Cholecystitis

Xanthogranulomatous Osteomyelitis of the Tibia

Xanthogranulomatous osteomyelitis of the humerus in a pediatric patient with Alagille syndrome: a case report and literature review

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