Osteofibrous dysplasia in the newborn. Report of a case.

Anderson, McCall; Townsend, David; Johnston, James O.; Bohay, D R

doi:10.2106/00004623-199302000-00014

Cited by 28 publications

(2 citation statements)

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“…Neonatal presentation with both unilateral and bilateral tibial disease has been previously described [13][14][15][16] . Clinically, the lesions were noted in childhood, with four of the six patients presenting before the age of ten years.…”

Section: Discussionmentioning

confidence: 99%

Familial Osteofibrous Dysplasia<sbt aid="964518">A Case Series</sbt>

Karol

Brown

Wise

et al. 2005

J Bone Joint Surg Am

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steofibrous dysplasia is a benign bone dysplasia in which lytic lesions occur in the cortex of the tibia and/or fibula in skeletally immature patients. Campanacci proposed the term "osteofibrous dysplasia" and described the clinical and pathologic findings 1,2 . The dysplasia may be clinically silent, so the true incidence of the disease is unknown. Symptomatic children present with anterolateral bowing of the affected bones or with pathologic fractures. As a result of the site of involvement and the propensity for pathologic fractures, this condition may be confused with congenital pseudarthrosis of the tibia.Pathologic specimens from sites affected by osteofibrous dysplasia show bland fibroblastic tissue associated with immature woven bone rimmed by osteoblasts 3-5 . While there has been a questionable association with evolution into adamantinoma, the pathologic appearance of osteofibrous dysplasia does not include neoplastic features.Osteofibrous dysplasia was not thought to be an inherited condition until a recent report, by Hunter and Jarvis, on two siblings with pathologic fractures through lesions resembling osteofibrous dysplasia 6 . We describe six individu-als, in three generations of a kindred of nineteen people, who had lytic lesions of the tibia that resembled osteofibrous dysplasia radiographically and histologically. All six patients were treated for pathologic fracture of the tibia or fibula at our institution. Materials and Methodsith institutional review board approval, a four-generation pedigree was obtained (Fig. 1). One patient presented with tibial lesions and gave a positive family history, after which medical records and radiographs were reviewed retrospectively to identify all of that patients' family members who had had lower-extremity fractures in childhood. All of the affected patients had been treated at our hospital between 1957 and the time of the study. There were twenty-two relatives from four generations; three had died before the initiation of the study.Radiographs of the tibia and fibula of the six affected patients were reviewed, and anteroposterior and lateral radiographs of the tibia and fibula of ten unaffected family members were made at the time of the study. These ten individuals O W Fig. 1 Pedigree of the family. Patients with documented tibial lesions in childhood are represented by numbers, family members with radiographic evidence of involvement are represented by shaded circles (females) or squares (males), family members with subtle radiographic findings are represented by partially shaded circles or squares, members who died before the time of the study are noted by a diagonal line. An asterisk indicates that a blood sample was obtained for DNA analysis, and a plus sign indicates that anteroposterior and lateral radiographs of the extremities were made.

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Section: Discussionmentioning

confidence: 99%

Familial Osteofibrous Dysplasia<sbt aid="964518">A Case Series</sbt>

Karol

Brown

Wise

et al. 2005

J Bone Joint Surg Am

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“…The age of presentation ranging from 7 days to 22 years [5] . However, in the literature, only few cases have been described in the neonatal period [ 6 – 10 ]. Diagnosis of congenital OFD is challenging because it is rare and the radiological findings are often nonspecific as they can mimic other neoplastic or nonneoplastic bone lesions.…”

Section: Introductionmentioning

confidence: 99%

Osteofibrous dysplasia: A rare case in 3-day-old female

Castaldo¹,

Siervo²,

Ferrara

et al. 2022

Radiology Case Reports

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Osteofibrous dysplasia (OFD) is a nonneoplastic tumor-like lesion, made up of fibrous matrix with immature bone tissue surrounded by osteoblasts, occurring usually in the cortex of tibial diaphysis. OFD is usually seen in the first decade of life and, according to literature, it is rarely seen in the newborn period. Diagnosis of congenital OFD in the newborn is challenging because it is uncommon in this age group and can be confused with other bone benign or malignant lesions. Imaging plays an important role in diagnosis, although histological confirmation is often required. Our report presents a rare case of pathologically confirmed congenital OFD in 3-day-old female which presented with a swelling of her right leg. We will focus on imaging findings of OFD and main differential diagnosis of this lesion in neonatal age.

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Tumors and Tumorlike Conditions of Bone

2002

Tumors of the Fetus and Infant

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Osteofibrous dysplasia in the newborn. Report of a case.

Cited by 28 publications

References 1 publication

Familial Osteofibrous Dysplasia<sbt aid="964518">A Case Series</sbt>

Familial Osteofibrous Dysplasia<sbt aid="964518">A Case Series</sbt>

Osteofibrous dysplasia: A rare case in 3-day-old female

Tumors and Tumorlike Conditions of Bone

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