The enigma of xanthogranulomatous osteomyelitis: report of a rare case with review of literature
DOI:
https://doi.org/10.18203/issn.2455-4510.IntJResOrthop20250467Keywords:
Osteomyelitis, Foamy Macrophages, Bone lesions, Neoplastic conditionAbstract
Xanthogranulomatous osteomyelitis (XO) is a rare chronic inflammatory condition histopathologically characterized by foamy macrophages, lymphocytes, plasma cells and fibrosis. It primarily affects organs like gallbladder and kidney but rarely involves bone. To date, only 21 cases of bone XO have been documented. XO mimics neoplastic bone lesions, creating diagnostic challenges necessitating a comprehensive approach that includes a thorough clinical history, detailed radiographic evaluation and definitive histopathologic examination. Hereby, we report a rare case of 25-year-old male presenting with pain, swelling and fever in the left forearm. Imaging revealed features suggestive of osteomyelitis and histopathological examination showed sheets of foamy macrophages, chronic inflammatory cells and fibrosis, thus clinching the diagnosis of XO. Through the case report we wish to highlight the importance of considering XO in the differential diagnosis of bone lesions so to avoid misdiagnosis of malignancy.
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References
Cozzutto C. Xanthogranulomatous osteomyelitis. Arch Pathol Lab Med. 1984;108(12):973-6.
Franco V, Aragona F, Genova G, Florena AM, Stella M, Campesi G. Xanthogranulomatous cholecystitis, Histopathological study and classification. Pathol Res Pract. 1990;186(3):383-90. DOI: https://doi.org/10.1016/S0344-0338(11)80297-9
Solooki S, Hoveidaei AH, Kardeh B, Azarpira N, Salehi E. Xanthogranulomatous Osteomyelitis of the Tibia. Ochsner J. 2019;19(3):276-81. DOI: https://doi.org/10.31486/toj.18.0165
Nakashiro H, Haraoka S, Fujiwara K, Harada S, Hisatsugu T, Watanabe T. Xanthogranulomatous cholecystitis. Cell composition and a possible pathogenetic role of cell-mediated immunity. Pathol Res Pract. 1995;191(11):1078-86. DOI: https://doi.org/10.1016/S0344-0338(11)80651-5
Kamat G, Gramapurohit V, Myageri A, Shettar C. Xanthogranulomatous osteomyelitis presenting as swelling in right tibia. Case Rep Pathol. 2011;257458. DOI: https://doi.org/10.1155/2011/257458
Vankalakunti M, Saikia UN, Mathew M, Mandeep K. Xanthogranulomatous osteomyelitis of ulna mimicking neoplasm. World J Surg Onc. 2007;5;46. DOI: https://doi.org/10.1186/1477-7819-5-46
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.
Mehdinezhad-Kashani M, Zarif-Zakerian B, Shayan K, Riyasi F. Xanthogranulomatous osteomyelitis of femur (report of one case). Iran J Orthop Surg. 2010;8(33):187-190.
Borjian A, Rezaei F, Eshaghi MA, Shemshaki H. Xanthogranulomatous osteomyelitis. J Orthop Traumatol. 2012;13(4):217-20. DOI: https://doi.org/10.1007/s10195-011-0165-8
Nunes R, Costa J, Martins M. Osteomielite xantogranulomatosa do úmero. Rev Port Ortop Traum. 2012;20(4):459-64.
Holmes BJ, Castelino-Prabhu S, Rosenthal DL, Ali SZ. Xanthogranuloma of bone: a challenging imitator of malignancy. Acta Cytol. 2013;57(2):198-202. DOI: https://doi.org/10.1159/000345900
Lee SH, Lee YH, Park H, Yong JC, Ho-Taek S, Woo IY, et al. A case report of xanthogranulomatous osteomyelitis of the distal ulna mimicking a malignant neoplasm. Am J Case Rep. 2013;14:304-7. DOI: https://doi.org/10.12659/AJCR.889422
Wang YL, Yu H, Zhang H, Cui XJ, Han JK. Xanthogranulomatous osteomyelitis of rib mimicking malignant lesions in (18)F-FDG PET/CT imaging: a report of two cases. Clin Nucl Med. 2014;39(5):e315-7. DOI: https://doi.org/10.1097/RLU.0b013e3182a20d45
Rathi M, Khattri J, Budania SK, Singh J, Awasthi S, Verma S. Xanthogranulomatous osteomyelitis. Arch Med Health Sci. 2014;2(2):228. DOI: https://doi.org/10.4103/2321-4848.144352
Nalini G. Xanthogranulomatous osteomyelitis: a case report. Med J. 2014;1(6):45-47.
Sapra R, Jain P, Gupta S, Kumar R. Multifocal bilateral xanthogranulomatous osteomyelitis. Indian J Orthop. 2015;49(4):482-4. DOI: https://doi.org/10.4103/0019-5413.159682
Singh S, Batra S, Maini L, Gautam VK. Xanthogranulomatous Osteomyelitis of Proximal Femur Masquerading as Benign Bone Tumor. Am J Orthop (Belle Mead NJ). 2015;44(8): E272-4.
Kaneuchi Y, Tajino T, Hakozaki M, Hitoshi Y, Kazuhiro T, Osamu H, et al. Xanthogranulomatous osteomyelitis of the distal tibia: a case report. Int J Rheum Dis. 2017;20(12):2238-41. DOI: https://doi.org/10.1111/1756-185X.12817
Arul P, Ramdas A, Varghese R, Kanchana B. Xanthogranulomatous osteomyelitis of femur masquerading as neoplasm. Clin Cancer Investig J. 2016;5(2):163. DOI: https://doi.org/10.4103/2278-0513.177134
Baisakh MR, Kar MR, Agrawal A, Mohapatra N. Xanthogranulomatous osteomyelitis mimicking neoplasm: a rare entity. Indian J Pathol Microbiol. 2016;59(2):253-4. DOI: https://doi.org/10.4103/0377-4929.182016
Cheema A, Arkader A, Pawel B. Xanthogranulomatous osteomyelitis of the humerus in a pediatric patient with Alagille syndrome: a case report and literature review. Skeletal Radiol. 2017;46(10):1447-52. DOI: https://doi.org/10.1007/s00256-017-2707-5
Kayser R, Mahlfeld K, Grasshoff H. Vertebral Langerhans cell histiocytosis in childhood-a differential diagnosis of spinal osteomyelitis. Klin Padiatr. 1999;211(5):399-402. DOI: https://doi.org/10.1055/s-2008-1043819
Amezyane T, Abouzahir A, Bassou D, Zoubeir Y, Hammi S, Mahassin F, et al. Pseudo-tumoral and ischemic encephalic Erdheim-Chester disease. Rev Neurol (Paris). 2009;165(6-7):591-5. DOI: https://doi.org/10.1016/j.neurol.2008.10.006
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(1):25-9. DOI: https://doi.org/10.1007/s002560050027
Kossard S, Chow E, Wilkinson B, Killingsworth M. Lipid and giant cell poor necrobiotic xanthogranuloma. J Cutan Pathol. 2000;27(7):374-8. DOI: https://doi.org/10.1034/j.1600-0560.2000.027007374.x
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(1):59-65. DOI: https://doi.org/10.1016/S0046-8177(99)90301-5