Recurrent Adamantinoma With Fibrous Dysplasia-like Feature

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) ha...

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Main Authors: Anja Petaros, Veljko Šantić, Anita Savić Vuković, Petar Perić, Nives Jonjić
Format: Article
Language:English
Published: SAGE Publishing 2024-04-01
Series:Clinical Pathology
Online Access:https://doi.org/10.1177/2632010X241240391
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author Anja Petaros
Veljko Šantić
Anita Savić Vuković
Petar Perić
Nives Jonjić
author_facet Anja Petaros
Veljko Šantić
Anita Savić Vuković
Petar Perić
Nives Jonjić
author_sort Anja Petaros
collection DOAJ
description 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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spelling doaj.art-a1207645cb6445ecae31a609595402052024-04-08T17:03:19ZengSAGE PublishingClinical Pathology2632-010X2024-04-011710.1177/2632010X241240391Recurrent Adamantinoma With Fibrous Dysplasia-like FeatureAnja Petaros0Veljko Šantić1Anita Savić Vuković2Petar Perić3Nives Jonjić4Department of Forensic Medicine and Criminalistics, Faculty of Medicine, University of Rijeka, Rijeka, CroatiaUniversity Hospital for Orthopaedic Surgery Lovran, Lovran, CroatiaDepartment of Pathology, Faculty of Medicine, University of Rijeka, Rijeka, CroatiaUniversity Hospital for Orthopaedic Surgery Lovran, Lovran, CroatiaDepartment of Pathology, Faculty of Medicine, University of Rijeka, Rijeka, CroatiaAdamantinoma (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.https://doi.org/10.1177/2632010X241240391
spellingShingle Anja Petaros
Veljko Šantić
Anita Savić Vuković
Petar Perić
Nives Jonjić
Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
Clinical Pathology
title Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
title_full Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
title_fullStr Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
title_full_unstemmed Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
title_short Recurrent Adamantinoma With Fibrous Dysplasia-like Feature
title_sort recurrent adamantinoma with fibrous dysplasia like feature
url https://doi.org/10.1177/2632010X241240391
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