Osteofibrous dysplasia-like adamantinoma versus osteofibrous dysplasia in children: A case report of challenging diagnosis

Kamal, Achmad Fauzi; Anshori, Fahmi; Kodrat, Evelina

doi:10.1016/j.ijscr.2021.01.093

Cited by 6 publications

(9 citation statements)

References 9 publications

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“…Complete involvement of the medullary cavity is almost always seen in an adamantinoma. In contrast to OFD and OFD-like adamantinoma, intra-medullary involvement is minimal or absent [32] . Consequently, treatment of patients with a diagnosis of OFD or differentiated adamantinoma tends to be conservative while patients with classic adamantinoma are treated more aggressively.…”

Section: Discussionmentioning

confidence: 91%

A 15 year-old-girl with persistent leg pain diagnosed as a giant cell tumor of the tibial diaphysis: A case report and review of the literature

Masud

Yadav²,

Yadav³

et al. 2022

International Journal of Surgery Case Reports

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Section: Discussionmentioning

confidence: 91%

A 15 year-old-girl with persistent leg pain diagnosed as a giant cell tumor of the tibial diaphysis: A case report and review of the literature

Masud

Yadav²,

Yadav³

et al. 2022

International Journal of Surgery Case Reports

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“…This supports the suggestion to rely on multiple biopsies for diagnosing AD, since a single biopsy with one tumor’s component predominating over the other can hinder the accurate diagnosis. Any (osteo)fibrous lesion accompanied by symptoms that can be linked to AD should be evaluated immunohistochemically, despite the apparently clear histology, 33 and not only with epithelial markers but also with podoplanin, since cytokeratins and EMA can show variable staining in the spindle cells of FD-like AD. This is supported by the fact that AD is a biphasic tumors that contain epithelial and mesenchymal component, such as synovial sarcoma, mesothelioma and ameloblastoma that also express podoplanin.…”

Section: Discussionmentioning

confidence: 99%

Recurrent Adamantinoma With Fibrous Dysplasia-like Feature

Petaros,

Šantić,

Vuković

et al. 2024

Clin Med�Insights�Pathol

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Adamantinoma (AD) is a rare, slow-growing primary malignant bone tumor characterized by a biphasic morphology of clusters of epithelial cells and spindle cell osteofibrous components. A strong relationship between AD and osteofibrous dysplasia (OFD) has been proposed, while fibrous dysplasia (FD) has been rarely associated with AD. We present an AD case that was followed and histologically evaluated 3 times over 6 years with different morphological patterns. The tumor in the primary biopsy and after complete resection showed classical features of AD and osteofibrous-like pattern, while the recurrent lesion presented with exclusively spindle cell morphology and was thus diagnosed as FD. However, the extensive immunohistochemical analysis in all 3 lesions revealed strong reactivity for pancytokeratin, vimentin, p63, and podoplanin, which are characteristic for AD. Although, in the FD-like section of the tumor from the first recurrence the positivity of podoplanin was stronger than pancitokeratin, which was variably positive on spindle cells. The present case highlights the problem of diagnosing AD based on a single biopsy with one tumor’s component predominating over the other, and at the same time emphasizes the importance of using immunohistochemical staining for keratin and podoplanin when the histopathological features of (osteo)fibrous lesion can be linked to AD.

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“…‘En bloc resection with wide margin of the OFD/AD, and surveillance of the OFD’ [ 11–13 ] considering OFD/AD as a histological subtype of AD [ 4 , 12 , 13 ]. A recent multicenter study proposed to reclassify OFD/AD as an aggressive local intermediate and not as a variant of adamantinoma [ 11 ].…”

Section: Discussionmentioning

confidence: 99%

Osteofibrous dysplasia-like adamantinoma of isolated fibula in a child mimicking chronic osteomyelitis with pathological fracture

Goetz

Dumitriu

Galant

et al. 2022

Journal of Surgical Case Reports

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The occurrence of a pathological fracture in children requires a rigorous diagnostic approach in order to establish the etiology and to develop a precise therapeutic strategy. Several causes are associated with these fractures, the most frequent being benign tumors in children in developed countries and chronic osteomyelitis in developing countries. More rarely, malignant tumors must however always be considered. The differential diagnosis on imaging may be difficult to establish between bone tumors and chronic infection. Surgical biopsy is therefore often performed to establish the precise origin of the fracture. We report the case of an adamantinoma (osteofibrous dysplasia-like) of the fibula in a 7-year-old child, discovered during the management of a pathologic fracture. The presumed diagnosis before biopsy was chronic osteomyelitis. A 14-cm-resection of the affected fibula was performed with good functional result. Differential diagnosis between adamantinoma, osteofibrous dysplasia and osteofibrous dysplasia-like adamantinoma remains very challenging.

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Osteofibrous dysplasia-like adamantinoma versus osteofibrous dysplasia in children: A case report of challenging diagnosis

Cited by 6 publications

References 9 publications

A 15 year-old-girl with persistent leg pain diagnosed as a giant cell tumor of the tibial diaphysis: A case report and review of the literature

A 15 year-old-girl with persistent leg pain diagnosed as a giant cell tumor of the tibial diaphysis: A case report and review of the literature

Recurrent Adamantinoma With Fibrous Dysplasia-like Feature

Osteofibrous dysplasia-like adamantinoma of isolated fibula in a child mimicking chronic osteomyelitis with pathological fracture

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