Background: Chronic Non-Infectious Osteomyelitis (CNO) is a chronic, relapsing, self-limiting inflammation of the bone. Although it is rare, CNO has been associated with inflammatory bowel disease and frequently precedes the initial diagnosis. We present a case of CNO in a patient with known ulcerative colitis in clinical remission who presented with purulent multifocal joint effusions in the setting of elevated inflammatory markers and fever suspicious for bacterial osteomyelitis. Case presentation: Our patient is a 12-year-old girl with ulcerative colitis who presented with fevers and insidious onset of joint pain at multiple sites. She had multiple joint effusions on imaging and blood and joint cultures were negative. Biopsy of the left acromion demonstrated acute and chronic osteomyelitis with areas of necrosis and granulomatous inflammation suggestive of CNO. Patient was started on high dose corticosteroids as well as methotrexate injections with marked improvement in symptoms. Conclusion: This case highlights that while purulent effusions are often indicative of bacterial osteomyelitis, the consideration of CNO in a patient with inflammatory bowel disease (IBD) with multifocal small bone involvement and negative blood cultures should be considered.
Primary osteosarcomas of the jaw (OSJ) are rare, accounting for 6% of all osteosarcomas. This study aims to determine the value of SATB2 and MDM2 immunohistochemistry (IHC) in differentiating OSJ from other jawbone mimickers, such as benign fibro-osseous lesions (BFOLs) of the jaw or Ewing sarcoma of the jaw. Certain subsets of osteosarcoma harbor a supernumerary ring and/or giant marker chromosomes with amplification of the 12q13–15 region, including the murine double-minute type 2 (MDM2) and cyclin-dependent kinase 4 (CDK4) genes. Special AT-rich sequence-binding protein 2 (SATB2) is an immunophenotypic marker for osteoblastic differentiation. Cases of OSJ, BFOLs (ossifying fibroma and fibrous dysplasia) of the jaw, and Ewing sarcoma of the jaw were retrieved from the Departments of Oral Pathology and Oral Medicine, Faculty of Dentistry, Obafemi Awolowo University and Lagos State University College of Medicine, Nigeria. All OSJ retrieved showed histologic features of high-grade osteosarcoma. IHC for SATB2 (clone EP281) and MDM2 (clone IF2), as well as fluorescence in situ hybridization (FISH) for MDM2 amplification, were performed on all cases. SATB2 was expressed in a strong intensity and diffuse staining pattern in all cases (11 OSJ, including a small-cell variant, 7 ossifying fibromas, and 5 fibrous dysplasias) except in Ewing sarcoma, where it was negative in neoplastic cells. MDM2 was expressed in a weak to moderate intensity and scattered focal to limited diffuse staining pattern in 27% (3/11) of cases of OSJ and negative in all BFOLs and the Ewing sarcoma. MDM2 amplification was negative by FISH in interpretable cases. In conclusion, the three cases of high-grade OSJs that expressed MDM2 may have undergone transformation from a low-grade osteosarcoma (LGOS). SATB2 is not a dependable diagnostic marker to differentiate OSJ from BFOLs of the jaw; however, it could serve as a valuable diagnostic marker in differentiating the small-cell variant of OSJ from Ewing sarcoma of the jaw, while MDM2 may be a useful diagnostic marker in differentiating OSJ from BFOLs of the jaw, especially in the case of an LGOS or high-grade transformed osteosarcoma.
Epithelioid hemangioendothelioma (EHE) is a rare vascular tumor with metastatic potential and estimated prevalence of less than one case per million. Among the musculoskeletal system, the long bones are commonly involved with approximately half patients experiencing multicentric involvement. Clinical course of EHE is often variable and nonspecific. Poorly demarcated osteolytic lesions are most commonly seen radiologically. Diagnostic confirmation is usually obtained by biopsy and histopathological exam, including immunostaining for endothelial markers. We present a rare case of unicentric EHE involving the calcaneum. Our patient had an indolent course of disease after surgical resection and no recurrence in seven years on clinical and radiological surveillance.
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