Summary
The most controversial aspect of osteofibrous dysplasia (OFD) is its possible histogenetic relationship to adamantinoma of long bone. Evidence is recently beginning to accumulate that OFD may be a reactive process to regressive adamantinoma. To verify the concept, 13 lesions of OFD were studied again by immunohistochemistry for cytokeratins of different molecular masses, as well as by conventional stainings. In addition, 2 adamantinomas and 6 fibrous dysplasias of the tibia were studied for reference. A small number of spindle- or ovoid-shaped cells scattered individually in the fibro-osseous stroma showed positive reactions for cytokeratins of 55–57 kDa in 2 lesions, and for those of 45–56.5 kDa in 8 lesions of 13 OFDs, although no definite epithelial island could be detected even by immunohistochemistry. Adamantinomas also showed single cytokeratin-positive cells dispersed in fibroblastic stroma, in addition to epithelial islands positive for cytokeratins of both 55–57 kDa and 45–56,5 kDa. All cases of fibrous dysplasia were negative for cytokeratins. During the observation, no case of OFDs progressed to classic adamantinoma. The present study, demonstrating the existence of an intermediate stage between “differentiated adamantinoma” and total elimination of adamantinomatous components, gives further support for the concept that OFD is a secondary reactive process to adamantinomatous tissue. In practice, the existence of single scattered cytokeratin-immunoreactive cells in otherwise typical OFDs may not indicate the truly malignant behaviour of classic adamantinoma, unless discrete epithelioid cell nests are also found.
Similar content being viewed by others
Abbreviations
- OFD:
-
osteofibrous dysplasia
References
Campanacci M (1976) Osteofibrous dysplasia of long bones. A new clinical entity. Ital J Orthop Traumatol 2:221–237
Campanacci M, Laus M (1981) Osteofibrous dysplasia of the tibia and fibula. J Bone Joint Surg 63:367–375
Cohen DM, Dahlin DC, Pugh DG (1962) Fibrous dysplasia associated with adamantinoma of the long bones. Cancer 15:515–521
Cordell JL, Falini B, Erber WN, Ghosh AK, Abdulaziz Z, Mac-Donald S, Pulford KAF, Stein H, Masson DY (1984) Immunoenzymatic labeling of monoclonal antibodies using immune complexes of alkaline phosphatase and monoclonal anti-alka-line phosphatase (APAAP complexes). J Histochem Cytochem 32:219–229
Czerniak B, Rojas-Corona RR, Dorfman HD (1989) Morphological diversity of long bone adamantinoma. The concept of differentiated (regressing) adamantinoma and its relationship to osteofibrous dysplasia. Cancer 64:2319–2334
Czernobilsky B, Moll R, Levy R, Franke WW (1985) Co-expression of cytokeratin and vimentin filaments in mesothelial, granulosa and rete ovarii cells of the human ovary. Eur J Cell Biol 37:175–190
Franke WW, Moll R (1987) Cytoskeletal components of lymphoid organs. I. Synthesis of cytokeratins 8 and 18 and desmin in subpopulations of extrafollicular reticulum cells of human lymph nodes, tonsils and spleen. Differentiation 36:145–163
Gown AM, Boyd HC, Chang Y, Ferguson M, Reichler B, Tippens D (1988) Smooth muscle cells can express cytokeratins of “simple” epithelium. Immunohistochemical and biochemical studies in vitro and in vivo. Am J Pathol 132:223–232
Jahn L, Fouquet B, Rohe K, Franke WW (1987) Cytokeratins in certain endothelial and smooth muscle cells of two taxonomically distinct vertebrate species,Xenopus laevis and man. Differentiation 36:234–254
Johnson LC (1972) Congenital pseudoarthrosis, adamantinoma of bone, and intracortical fibrous dysplasia of the tibia. J Bone Joint Surg 54:1355
Keeney GL, Unni KK, Beabout JW, Pritchard DJ (1989) Adamantinoma of long bones. Clinicopathologic study of 85 cases. Cancer 64:730–737
Kempson RL (1966) Ossifying fibroma of long bones. Arch Pathol 82:218–233
Markel SF (1978) Ossifying fibroma of long bone: its distinction from fibrous dysplasia and its association with adamantinoma of long bone. Am J Clin Pathol 69:91–97
Miettinen M, Virtanen I, Talerman A (1985) Intermediate filament proteins in human testis and testicular germ-cell tumors. Am J Pathol 120:402–410
Mirra JM (1989) Adamantinoma and osteofibrous dysplasia. In: Mirra JM (ed) Bone tumors. Clinical, radiologic, and pathologic correlations, vol II. Lee & Febiger, Philadelphia, pp 1203–1231
Muijen GNP van, Ruiter DJ, Warnaar SO (1987) Coexpression of intermediate filament polypeptides in human fetal and adult tissues. Lab Invest 57:359–369
Perez-Atayde AR, Kozakewich HPW, Vawter GF (1985) Adamantinoma of the tibia. An ultrastructural and immunohistochemical study. Cancer 55:1015–1023
Rosai J, Pinkus GS (1982) Immunohistochemical demonstration of epithelial differentiation in adamantinoma of the tibia. Am J Surg Pathol 6:427–434
Turley H, Pulford KAF, Gatter KC, Mason DY (1988) Biochemical evidence that cytokeratins are present in smooth muscle. Br J Exp Pathol 69:433–440
Ueda Y, Bosse A, Roessner A, Edel G, Wuisman P, Böcker W (1991) Juvenile intracortical adamantinoma of the tibia with predominant osteofibrous dysplasia-like features. Pathol Res Pract (in press)
Unni KK, Dahlin DC, Beabout JW, Ivins JC (1974) Adamantinoma of long bones. Cancer 34:1796–1805
Weiss SW, Dorfman HD (1977) Adamantinoma of long bone: an analysis of nine new cases with emphasis on metastasizing lesions and fibrous dysplasia-like changes. Hum Pathol 8:141–153
Author information
Authors and Affiliations
Additional information
Dedicated to Professor E. Grundmann on the occasion of his 70th birthday
Rights and permissions
About this article
Cite this article
Ueda, Y., Blasius, S., Edel, G. et al. Osteofibrous dysplasia of long bones—a reactive process to adamantinomatous tissue. J Cancer Res Clin Oncol 118, 152–156 (1992). https://doi.org/10.1007/BF01187505
Received:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/BF01187505