Neonatal osteofibrous dysplasia: Synchronous tibial and fibular involvement is common.


Journal

BJR case reports
ISSN: 2055-7159
Titre abrégé: BJR Case Rep
Pays: England
ID NLM: 101684132

Informations de publication

Date de publication:
Sep 2022
Historique:
received: 07 04 2021
revised: 30 07 2021
accepted: 09 01 2022
entrez: 14 9 2022
pubmed: 15 9 2022
medline: 15 9 2022
Statut: epublish

Résumé

Osteofibrous dysplasia is a rare non-malignant fibro-osseous bone tumour, first described and characterised under this name by Campanacci in 1976. It is most commonly encountered in the tibia of children and young adults, but less frequently seen in the neonate with only few prior reports in the literature. We report a case of neonatal congenital osteofibrous dysplasia, presenting with unilateral limb deformity at birth. Radiographs demonstrated well-defined mixed lytic-sclerotic lesions, in a previously unreported distribution in this age-group, confined to the distal metadiaphysis of the affected tibia and fibula. Open surgery was performed for deformity correction, with tissue biopsy confirming the radiographically-suspected diagnosis. We present the up-to-date clinical, radiological and pathological findings in this case of a rare pathology with some novel features, within this age group, in disease distribution and consequent radiographic appearances. OFD should be considered in the differential of similar congenital deforming bone lesions of the lower limb. We also review the small number of previously published cases of congenital OFD in the neonate, noting in particular that the frequency of ipsilateral fibular involvement appears to be higher than that observed in older patients.

Identifiants

pubmed: 36101735
doi: 10.1259/bjrcr.20210072
pmc: PMC9461730
doi:

Types de publication

Case Reports

Langues

eng

Pagination

20210072

Informations de copyright

© 2022 The Authors. Published by the British Institute of Radiology.

Déclaration de conflit d'intérêts

Conflict of Interest: The authors declare that they have no conflict of interest. No relevant funding obtained for this manuscript.

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Auteurs

Harsimran Laidlow-Singh (H)

Department of Radiology, Broomfield Hospital, Chelmsford, United Kingdom.

Mark Latimer (M)

Academic Unit of Child Health, University of Sheffield, Sheffield, United Kingdom.

Pearl Wou (P)

Department of Radiology, Broomfield Hospital, Chelmsford, United Kingdom.

Amaka C Offiah (AC)

Academic Unit of Child Health, University of Sheffield, Sheffield, United Kingdom.

Classifications MeSH