An unusual case of adamantinoma of long bone

ABSTRACT Adamantinoma of the long bones is an exceedingly rare and slow-growing tumor that affects the diaphysis of long bones, particularly the tibia. Based on the pattern of the epithelial cell component and the presence or absence of the osteofibrous dysplasia-like element, several histological variants have been described, such as (i) tubular (the most frequent), (ii) basaloid, (iii) squamous, (iv) spindle variant, (v) osteofibrous dysplasia –like variant, and (vi) Ewing’s sarcoma – like adamantinoma (the least frequent). The diagnosis may be challenging since this tumor may be mistakenly interpreted as carcinoma, myoepithelial tumor, osteofibrous dysplasia, and vascular tumor. We report the case of a 41-year-old male who presented with swelling over the right leg associated with pain. The X-ray showed a lytic lesion of the right-sided tibia. The diagnosis of adamantinoma was made based on the clinico-radiological, histomorphology, and immunohistochemical findings. Histologically, classic adamantinoma is a biphasic tumor characterized by epithelial and osteofibrous components in varying proportions and differentiating patterns. The diagnosis can be confirmed by immunohistochemistry for demonstrating sparse epithelial cell nests when the radiological features are strongly consistent with adamantinoma. This case is highlighted because the epithelial component can lead to a misdiagnosis, particularly when the clinico-radiological features are overlooked. Adamantinoma of long bones has the potential for local recurrence and may metastasize to the lungs, lymph nodes, or other bones. The prognosis is good if early intervention is taken.

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Main Authors: Kumar,Arvind, Sharma,Ruchi, Verma,Anil Kumar, Tiwari,Abhijeet, Mishra,Jyoti
Format: Digital revista
Language:English
Published: Hospital Universitário da Universidade de São Paulo 2021
Online Access:http://old.scielo.br/scielo.php?script=sci_arttext&pid=S2236-19602021000100519
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spelling oai:scielo:S2236-196020210001005192021-05-26An unusual case of adamantinoma of long boneKumar,ArvindSharma,RuchiVerma,Anil KumarTiwari,AbhijeetMishra,Jyoti Adamantinoma Diaphyses Tibia ABSTRACT Adamantinoma of the long bones is an exceedingly rare and slow-growing tumor that affects the diaphysis of long bones, particularly the tibia. Based on the pattern of the epithelial cell component and the presence or absence of the osteofibrous dysplasia-like element, several histological variants have been described, such as (i) tubular (the most frequent), (ii) basaloid, (iii) squamous, (iv) spindle variant, (v) osteofibrous dysplasia –like variant, and (vi) Ewing’s sarcoma – like adamantinoma (the least frequent). The diagnosis may be challenging since this tumor may be mistakenly interpreted as carcinoma, myoepithelial tumor, osteofibrous dysplasia, and vascular tumor. We report the case of a 41-year-old male who presented with swelling over the right leg associated with pain. The X-ray showed a lytic lesion of the right-sided tibia. The diagnosis of adamantinoma was made based on the clinico-radiological, histomorphology, and immunohistochemical findings. Histologically, classic adamantinoma is a biphasic tumor characterized by epithelial and osteofibrous components in varying proportions and differentiating patterns. The diagnosis can be confirmed by immunohistochemistry for demonstrating sparse epithelial cell nests when the radiological features are strongly consistent with adamantinoma. This case is highlighted because the epithelial component can lead to a misdiagnosis, particularly when the clinico-radiological features are overlooked. Adamantinoma of long bones has the potential for local recurrence and may metastasize to the lungs, lymph nodes, or other bones. The prognosis is good if early intervention is taken.info:eu-repo/semantics/openAccessHospital Universitário da Universidade de São PauloAutopsy and Case Reports v.11 20212021-01-01info:eu-repo/semantics/reporttext/htmlhttp://old.scielo.br/scielo.php?script=sci_arttext&pid=S2236-19602021000100519en10.4322/acr.2021.276
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language English
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author Kumar,Arvind
Sharma,Ruchi
Verma,Anil Kumar
Tiwari,Abhijeet
Mishra,Jyoti
spellingShingle Kumar,Arvind
Sharma,Ruchi
Verma,Anil Kumar
Tiwari,Abhijeet
Mishra,Jyoti
An unusual case of adamantinoma of long bone
author_facet Kumar,Arvind
Sharma,Ruchi
Verma,Anil Kumar
Tiwari,Abhijeet
Mishra,Jyoti
author_sort Kumar,Arvind
title An unusual case of adamantinoma of long bone
title_short An unusual case of adamantinoma of long bone
title_full An unusual case of adamantinoma of long bone
title_fullStr An unusual case of adamantinoma of long bone
title_full_unstemmed An unusual case of adamantinoma of long bone
title_sort unusual case of adamantinoma of long bone
description ABSTRACT Adamantinoma of the long bones is an exceedingly rare and slow-growing tumor that affects the diaphysis of long bones, particularly the tibia. Based on the pattern of the epithelial cell component and the presence or absence of the osteofibrous dysplasia-like element, several histological variants have been described, such as (i) tubular (the most frequent), (ii) basaloid, (iii) squamous, (iv) spindle variant, (v) osteofibrous dysplasia –like variant, and (vi) Ewing’s sarcoma – like adamantinoma (the least frequent). The diagnosis may be challenging since this tumor may be mistakenly interpreted as carcinoma, myoepithelial tumor, osteofibrous dysplasia, and vascular tumor. We report the case of a 41-year-old male who presented with swelling over the right leg associated with pain. The X-ray showed a lytic lesion of the right-sided tibia. The diagnosis of adamantinoma was made based on the clinico-radiological, histomorphology, and immunohistochemical findings. Histologically, classic adamantinoma is a biphasic tumor characterized by epithelial and osteofibrous components in varying proportions and differentiating patterns. The diagnosis can be confirmed by immunohistochemistry for demonstrating sparse epithelial cell nests when the radiological features are strongly consistent with adamantinoma. This case is highlighted because the epithelial component can lead to a misdiagnosis, particularly when the clinico-radiological features are overlooked. Adamantinoma of long bones has the potential for local recurrence and may metastasize to the lungs, lymph nodes, or other bones. The prognosis is good if early intervention is taken.
publisher Hospital Universitário da Universidade de São Paulo
publishDate 2021
url http://old.scielo.br/scielo.php?script=sci_arttext&pid=S2236-19602021000100519
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