TY - JOUR
T1 - Osteofibrous dysplasia-like adamantinoma versus osteofibrous dysplasia in children
T2 - A case report of challenging diagnosis
AU - Kamal, Achmad Fauzi
AU - Anshori, Fahmi
AU - Kodrat, Evelina
N1 - Funding Information:
The relationship of osteofibrous dysplasia with adamantinoma is unclear. Czerniak et al. [ 3 ] reported that there is a continuum of lesions with classic adamantinoma at one end and osteofibrous dysplasia at the other. This hypothesis is also supported by another author considered this possible relationship by calling osteofibrous dysplasia as “juvenile adamantinoma”. This hypothesis, however does not set aside the possibility of de novo osteofibrous dysplasia that has no relation to adamantinoma. However, the study determined that there was no definite evidence of a precursor role for osteofibrous dysplasia [ 3 , 9 , 11 ].
Publisher Copyright:
© 2021 The Authors
Copyright:
Copyright 2021 Elsevier B.V., All rights reserved.
PY - 2021/3
Y1 - 2021/3
N2 - Introduction: Osteofibrous dysplasia (OFD) and Osteofibrous dysplasia-like Adamantinoma have a similar appearance both in clinical and radiography, but different in its histopathology. Despite this similarity, the treatment and prognosis are different, therefore the diagnosis should be established precisely. Case illustration: A three-year-old boy was admitted to hospital after falling on his lower leg. A bead size lump appeared on his tibia with pain and swelling, which later became enlarged. Diagnosis of osteofibrous dysplasia and adamantinoma was considered. We performed limb-salvage procedure by curretage, bone grafting, and internal fixation application. The histology section showed woven bone rimmed by polygonal osteoblast cell with intervening fibrous stroma and small nests of tumour cells raised the possibility of epithelial differentiation. The positivity for cytokeratin immunostaining confirmed the diagnosis as osteofibrous dysplasia-like adamantinoma. In this case it is a very rare spectrum of malignancy in children. Discussion: These two tumor entities have identical radiographic characteristics, histopathology features the distinction between classic adamantinoma and OFD-like adamantinoma based on the predominant epithelial component. The relationship of osteofibrous dysplasia with adamantinoma is unclear. Several authors considered possible calling relationship osteofibrous dysplasia as “juvenile adamantinoma”. However, does not rule out the possible existence of de novo osteofibrous dysplasia not related to adamantinoma. Conclusions: OFD-like adamantinoma and Osteofibrous Dysplasia had similar histopathology pattern, a pathologist must be aware of this feature and perform immunohistochemical staining for keratin particularly when the histopathological feature of osteofibrous dysplasia showed small nests of tumor cells within the fibrous stroma. diagnostic challenging and require multidisciplinary approach.
AB - Introduction: Osteofibrous dysplasia (OFD) and Osteofibrous dysplasia-like Adamantinoma have a similar appearance both in clinical and radiography, but different in its histopathology. Despite this similarity, the treatment and prognosis are different, therefore the diagnosis should be established precisely. Case illustration: A three-year-old boy was admitted to hospital after falling on his lower leg. A bead size lump appeared on his tibia with pain and swelling, which later became enlarged. Diagnosis of osteofibrous dysplasia and adamantinoma was considered. We performed limb-salvage procedure by curretage, bone grafting, and internal fixation application. The histology section showed woven bone rimmed by polygonal osteoblast cell with intervening fibrous stroma and small nests of tumour cells raised the possibility of epithelial differentiation. The positivity for cytokeratin immunostaining confirmed the diagnosis as osteofibrous dysplasia-like adamantinoma. In this case it is a very rare spectrum of malignancy in children. Discussion: These two tumor entities have identical radiographic characteristics, histopathology features the distinction between classic adamantinoma and OFD-like adamantinoma based on the predominant epithelial component. The relationship of osteofibrous dysplasia with adamantinoma is unclear. Several authors considered possible calling relationship osteofibrous dysplasia as “juvenile adamantinoma”. However, does not rule out the possible existence of de novo osteofibrous dysplasia not related to adamantinoma. Conclusions: OFD-like adamantinoma and Osteofibrous Dysplasia had similar histopathology pattern, a pathologist must be aware of this feature and perform immunohistochemical staining for keratin particularly when the histopathological feature of osteofibrous dysplasia showed small nests of tumor cells within the fibrous stroma. diagnostic challenging and require multidisciplinary approach.
KW - Immunohistochemical staining
KW - Osteofibrous dysplasia
KW - Osteofibrous dysplasia-like adamantinoma
UR - http://www.scopus.com/inward/record.url?scp=85101451168&partnerID=8YFLogxK
U2 - 10.1016/j.ijscr.2021.01.093
DO - 10.1016/j.ijscr.2021.01.093
M3 - Article
AN - SCOPUS:85101451168
SN - 2210-2612
VL - 80
JO - International Journal of Surgery Case Reports
JF - International Journal of Surgery Case Reports
M1 - 105599
ER -