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The Journal of Bone and Joint Surgery, Vol 76, Issue 10 1482-1499, Copyright © 1994 by Journal of Bone and Joint Surgery, Inc
Adamantinoma of the long bones. A clinicopathological study of thirty-two patients with emphasis on histological subtype, precursor lesion, and biological behavior
HM Hazelbag, AH Taminiau, GJ Fleuren and PC Hogendoorn
Department of Pathology, University of Leiden, The Netherlands.
The records of thirty-two patients who had had an adamantinoma of the long
bones were examined to investigate the relationship between the clinical
presentation, the histological subtype, and the method of treatment, and
the clinical result. All histological patterns of differentiation that are
characteristic of adamantinoma were observed, including the basaloid,
spindle-cell, tubular, squamous, and osteofibrous dysplasia-like subtypes.
Follow-up data were available for twenty-eight (88 per cent) of the
thirty-two patients. These patients were followed for a mean duration of
122 months (range, eleven months to twenty-nine years and two months). Nine
patients (32 per cent), all of whom had been managed with an intralesional
or marginal procedure, had a local recurrence of the tumor after a mean
disease developed in three of the nine patients. In five other patients,
metastasis developed without having been preceded by a local recurrence.
Thus, the over-all rate of metastasis was 29 per cent (eight patients). The
mean duration of survival for the patients who had metastasis was twelve
years and eight months. Statistical analysis of various clinicopathological
variables revealed intralesional or marginal excision to be the most
significant risk factor for a local recurrence or metastasis (p <
0.001). Two patients who had had a presumed osteofibrous dysplasia-like
adamantinoma, which contained few isolated keratin-positive epithelial
cells within the stroma at the time of presentation, had a full-blown
adamantinoma at the time of the local recurrence. Although the clinical
course that was observed may be the result of a sampling error, it poses
questions as to the regressive nature of osteofibrous dysplasia-like
adamantinoma. On the basis of our findings and the data in the literature,
we believe that an osteofibrous dysplasia-like adamantinoma may be a
precursor lesion of the classic type of adamantinoma.
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