Skip to main content

Advertisement

SpringerLink
Log in

Adamantinoma, osteofibrous dysplasia and differentiated adamantinoma

  • Review Article
  • Published:
Skeletal Radiology Aims and scope Submit manuscript

Abstract

For just over 100 years, adamantinoma has been recognized as a primary bone tumor with epithelial characteristics and predominantly involving the tibia. Osteofibrous dysplasia is a fibro-osseous lesion also predominantly confined to the tibia with radiologic features similar to those of adamantinoma. This lesion has been shown by immunohistochemical studies to frequently contain cytokeratin-positive epithelial cells. More recently, a third group of cases with clinical and radiologic features similar to those of osteofibrous dysplasia have demonstrated more overt strands of epithelial cells within a fibro-osseous background and have been categorized as "differentiated", "regressive", "juvenile intracortical" or "osteofibrous dysplasia-like" adamantinoma. Cytokeratin subset immunohistochemical stains and cytogenetic studies performed in recent years suggest a common histogenesis for these three entities. This article reviews the clinical, radiologic and pathologic features of these entities as well as their prognostic significance. It also reviews the results of the immunohistochemical and cytogenetic studies which establish a common histogenetic relationship.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1.
Fig. 2. A
Fig. 3.
Fig. 4.
Fig. 5.
Fig. 6. A
Fig. 7.
Fig. 8.
Fig. 9. A
Fig. 10. A
Fig. 11A–D.
Fig. 12A, B.
Fig. 13A–C.
Fig. 14A–C.
Fig. 15. A
Fig. 16. A
Fig. 17. A, B

Similar content being viewed by others

References

  1. Maier C. Ein primäres myelogenes platten epithelcarcinom der ulna. Bruns Beitr Klin Chir 1900; 26:553.

    Google Scholar 

  2. Fischer B. Über ein primäres Adamantinoma der Tibia. Virchows Arch [B] 1913; 12:422–441.

    Google Scholar 

  3. Schulenburg CAR. Adamantinoma. Ann R Coll Surg Engl 1951; 8:329–353.

    Google Scholar 

  4. Iravani S, Walling A, Gilbert-Barness E. Pathological case of the month. Arch Pediatr Adolesc Med 1997; 151:427–428.

    Google Scholar 

  5. Moon NF, Mori H. Adamantinoma of the appendicular skeleton—updated. Clin Orthop 1986; 204:215–237.

    PubMed  Google Scholar 

  6. Plump D, Haponik EF, Katz RS. Primary adamantinoma of the rib: thoracic manifestations of a rare bone tumor. South Med J 1986; 79:352–355.

    CAS  PubMed  Google Scholar 

  7. Mills SE, Rosai J. Adamantinoma of the pretibial soft tissue. Clinicopathologic features, differential diagnosis and possible relationship to intraosseous disease. Am J Clin Pathol 1985; 83:108–114.

    CAS  PubMed  Google Scholar 

  8. Bertoni F, Zucchi V, Mapelli S, Bacchini P. Case report 506: Adamantinoma of the soft tissues of leg. Skeletal Radiol 1988; 17:522–526.

    CAS  PubMed  Google Scholar 

  9. Altmannsberger M, Poppe H, Schauer A. An unusual case of adamantinoma of long bones. J Cancer Res Clin Oncol 1982; 104:315–320.

    CAS  PubMed  Google Scholar 

  10. Van Schoor JX, Vallaeys JH, Joos GF, Roels HJ, Pauwels RA, Van der Straeten ME. Adamantinoma of the tibia with pulmonary metastases and hypercalcemia. Chest 1991; 100:279–281.

    PubMed  Google Scholar 

  11. Bohndorf K, Nidecker A, Mathias K, Zidkova H, Kauffmann H, Jundt G. The radiologic findings in adamantinoma of the long tubular bones. Rofo Fortschr Geb Rontgenstrahl Neuen Bildgeb Verfahr 1992; 157:239–244.

    CAS  Google Scholar 

  12. Young JW, Aisner SC, Resnick CS, Levine AM, Dorfman HD, Whitley NO. Case report 660: Adamantinoma of tibia. Skeletal Radiol 1991; 20:152–156.

    CAS  PubMed  Google Scholar 

  13. Rosai J, Pincus GS. Immunohistochemical demonstration of epithelial differentiation in adamantinoma of the tibia. Am J Surg Pathol 1982; 6:427–434.

    CAS  PubMed  Google Scholar 

  14. Ryrie BJ. Adamantinoma of the tibia: etiology and pathogenesis. BMJ 1932; ii:1000–1003.

    Google Scholar 

  15. Lipper S, Kahn LB. Case report 235:. Ewing-like adamantinoma of the left radial head and neck. Skeletal Radiol 1983; 10:61–66.

    CAS  PubMed  Google Scholar 

  16. Ishida T, Kikuchi F, Oka T, Machinami R, Kojima T, Iijima T, Higaki S, Imamura T. Case report 727: Juxtacortical adamantinoma of humerus (simulating Ewing tumor). Skeletal Radiol 1992; 21:205–209.

    CAS  PubMed  Google Scholar 

  17. Fukunaga M, Ushigome S. Periosteal Ewing-like adamantinoma. Virchows Arch 1998; 433:385–389.

    Article  CAS  PubMed  Google Scholar 

  18. Bridge JA, Fuller ME, Neff JR, Degenhardt J, Wang M, Walker C, Dorfman HD, Baker KS, Seemayer TA. Adamantinoma-like Ewing's sarcoma: genomic confirmation, phenotypic drift. Am J Surg Pathol 2000; 24:322–323.

    Article  PubMed  Google Scholar 

  19. Hauben E, van den Broek LC, Van Marck E, Hogendoorn PC. Adamantinoma-like Ewing's sarcoma and Ewing's-like adamantinoma. The T (11;22), T (21;22) status. J Pathol 2001; 195:218–221.

    Article  CAS  PubMed  Google Scholar 

  20. Galera-Davidson H, Fernandez-Rodriquez A, Torres-Olivera FJ, Gomez-Pascual A, Morena-Fernandez A. Cytologic diagnosis of a recurrent adamantinoma. Acta Cytol 1989; 33:635–638.

    CAS  PubMed  Google Scholar 

  21. Tabei SZ, Abdollahi B, Nili F. Diagnosis of metastatic adamantinoma of the tibia by pulmonary brushing cytology. Acta Cytol 1988; 32:579–581.

    CAS  PubMed  Google Scholar 

  22. Perez-Ordonez B, Bedard YC. Metastatic adamantinoma diagnosed by fine-needle aspiration biopsy of the lung. Diagn Cytopathol 1994; 10:347–351.

    CAS  PubMed  Google Scholar 

  23. Yoneyama T, Winter WG, Mislow L. Tibial adamantinoma: its histogenesis from ultrastructural studies. Cancer 1977; 40:1138–1142.

    CAS  PubMed  Google Scholar 

  24. Lombart-Bosch A, Ortuno-Pacheco G. Ultrastructural findings supporting the angioblastic nature of so-called adamantinoma of the tibia. Histopathology 1978; 2:189–200.

    PubMed  Google Scholar 

  25. Mori H, Shima R, Nakanishi H, Yoshida A, Fukunishi R. Adamantinoma of the tibia: a case report and a statistical review of reported cases. Acta Pathol Jpn 1981; 31:701–709.

    CAS  PubMed  Google Scholar 

  26. Knapp RH, Wick MR, Scheithauer BW, Unni KK. Adamantinoma of bone. An electron microscopic and immunohistochemical study. Virchows Arch [A] 1982; 398:75–86.

    Google Scholar 

  27. Pieterse AS, Smith PS, McClure J. Adamantinoma of long bones: clinical, pathological and ultrastructural features. J Clin Pathol 1982; 35:780–786.

    CAS  PubMed  Google Scholar 

  28. Mori H, Yamamoto S, Hiramatsu K, Miura T, Moon NF. Adamantinoma of the tibia. Ultrastructural and immunohistochemical study with reference to histogenesis. Clin Orthop 1984; 190:299–310.

    PubMed  Google Scholar 

  29. Jundt G, Remberger K, Roessner A, Schulz A, Bohndorf K. Adamantinoma of long bones. A histopathological and immunohistochemical study of 23 cases. Pathol Res Pract1995; 191:112–120.

  30. Hazelbag HM, Fleuren JG, van den Broek LJ, Taminiau AH, Hogendoorn PC. Adamantinoma of the long bones: keratin subclass immunoreactivity pattern with reference to its histogenesis. Am J Surg Pathol 1993; 17:1225–1233.

    CAS  PubMed  Google Scholar 

  31. Bovee JV, van den Broek LJ, de Boer WI, Hogendoorn PC. Expression of growth factors and their receptors in adamantinoma of long bones and the implications for its histogenesis. J Pathol 1998; 184:24–30.

    Article  CAS  PubMed  Google Scholar 

  32. Hazelbag HM, Fleuren GJ, Cornelisse CJ, van den Broek LJ, Taminiau AH, Hogendoorn PC. DNA aberrations in the epithelial cell component of adamantinoma of long bones. Am J Pathol 1995; 147:1770–1779.

    CAS  PubMed  Google Scholar 

  33. Hazelbag HM, Wessels JW, Mollevangers P, van den Berg E, Molenaar WM, Hogendoorn PC. Cytogenic analysis of adamantinoma of long bones: further indications for a common histogenesis with osteofibrous dysplasia. Cancer Genetic Cytogenet 1997; 97:5–11.

    Article  CAS  Google Scholar 

  34. Bridge JA, Dembinski A, DeBoer J, Travis J, Neff JR. Clonal chromosomal abnormalities in osteofibrous dysplasia: implications for histopathogenesis and its relationship with adamantinoma. Cancer 1994; 73:1746–1752.

    CAS  PubMed  Google Scholar 

  35. Kanamori M, Antonescu CR, Scott M, Bridge RS Jr, Neff JR, Spanier SS, Scarborough MT, Vergara G, Rosenthal HG, Bridge JA. Extra copies of chromosomes 7, 8, 12, 19 and 21 are recurrent in adamantinoma. J Mol Diagn 2001; 3:16–21.

    CAS  PubMed  Google Scholar 

  36. De Keyser F, Vansteenkiste J, Van Den Brande P, Demedts M, Van de Woestijne KP. Pulmonary metastasis of a tibial adamantinoma. Case report and review of the literature. Acta Clin Belg 1990; 45:31–33.

    PubMed  Google Scholar 

  37. Keeney GL, Unni KK, Beabout JW, Pritchard DJ. Adamantinoma of long bones. A clinicopathologic study of 85 cases. Cancer 1989; 64:730–737.

    CAS  PubMed  Google Scholar 

  38. Hazelbag HM, Taminiau AH, Fleuren GJ, Hogendoorn PCW. Adamantinoma of the long bones. A clinicopathologic study of thirty-two patients with emphasis on histological subtype, precursor lesion and biological behavior. J Bone Joint Surg Am 1994; 76:1482–1499.

    CAS  PubMed  Google Scholar 

  39. Qureshi AA, Shott S, Mallin BA, Gitelis S. Current trends in the management of adamantinoma of long bones. An international study. J Bone Joint Surg Am 2000; 82:1122–1131.

    Google Scholar 

  40. Campanacci M. Osteofibrous dysplasia of long bones, a new clinical study. Ital J Orthop Traumatol 1976; 2:221–237.

    CAS  PubMed  Google Scholar 

  41. Kempson RL. Ossifying fibroma of the long bones, a light and electron microscopic study. Arch Pathol Lab Med 1966; 82:218–233.

    CAS  Google Scholar 

  42. Johnson LC. Congenital pseudarthrosis, adamantinoma of the long bone and intracortical fibrous dysplasia of the tibia (abstract). Lab Invest 1973; 28:387.

    Google Scholar 

  43. Semian DW, Willis JB, Bove KE. Congenital fibrous defect of the tibia mimicking fibrous dysplasia. A case report. J Bone Joint Surg Am 1975; 57:854–857.

    CAS  PubMed  Google Scholar 

  44. Campanacci M, Laus M. Osteofibrous dysplasia of the tibia and fibula. J Bone Joint Surg Am 1981; 63:367–375.

    CAS  PubMed  Google Scholar 

  45. Park YK, Unni KK, McLeod RA, Pritchard DJ. Osteofibrous dysplasia: clinicopathologic study of 80 cases. Hum Pathol 1993; 24:1339–1347.

    CAS  PubMed  Google Scholar 

  46. Sweet DE, Vinh TN, Devaney K. Cortical osteofibrous dysplasia of long bones and its relationship to adamantinoma. A clinicopathologic study of 30 cases. Am J Surg Pathol 1992; 16:282–290.

    CAS  PubMed  Google Scholar 

  47. Ueda Y, Blasius S, Edel G, Wuisman P, Bocker W, Roessner A. Osteofibrous dysplasia of long bones—a reactive process to adamantinomatous tissue. J Cancer Res Clin Oncol 1992; 118:152–156.

    CAS  PubMed  Google Scholar 

  48. Ishida T, Iijma T, Kikuchi F, Kitagawa T, Tanida T, Imamura T, Machinami R. A clinicopathological and immunohistochemical study of osteofibrous dysplasia, differentiated adamantinoma and adamantinoma of long bones. Skeletal Radiol 1992; 21:493–502.

    CAS  PubMed  Google Scholar 

  49. Benassi MS, Campanacci L, Gamberi G, Ferrari C, Picci P, Sangiorgi L, Campanacci M. Cytokeratin expression and distribution in adamantinoma of the long bones and osteofibrous dysplasia of tibia and fibula. An immunohistochemical study correlated to histogenesis. Histopathology 1994; 25:71–76.

    CAS  PubMed  Google Scholar 

  50. Williams HK, Mangham C, Speight PM. Juvenile ossifying fibroma. An analysis of eight cases and a comparison with other fibro-osseous lesions. J Oral Pathol Med 2000; 29:13–18.

    Article  CAS  PubMed  Google Scholar 

  51. Ueda Y, Roessner A, Bosse A, Edel G, Bocker W, Wuisman P. Juvenile intracortical adamantinoma of the tibia with predominant osteofibrous dysplasia-like features. Pathol Res Pract 1991; 187:1039–1043.

    CAS  PubMed  Google Scholar 

  52. Czerniak B, Rojas-Corona RR, Dorfman HD. Morphologic diversity of long bone adamantinoma. The concept of differentiated (regressing) adamantinoma and its relationship to osteofibrous dysplasia. Cancer 1989; 64:19–34.

    Google Scholar 

  53. Kim JY, Kang GH, Chi JG. Adamantinoma of the tibia with predominant features of fibrous dysplasia : a case report. J Korean Med Sci 1996; 11:444–448.

    CAS  PubMed  Google Scholar 

  54. Konrad EA, Meister P, Stotz S. "Adamantinoma" of the tibia and reactive bone changes. Arch Orthop Trauma Surg 1978; 92:297–301.

    CAS  PubMed  Google Scholar 

  55. Schneider H, Enderle A. Differential diagnosis of a metastasizing adamantinoma of the tibia and fibula. Arch Orthop Trauma Surg 1979; 94:143–149.

    CAS  PubMed  Google Scholar 

  56. Unni KK, Dahlin DC, Beabout JW, Ivins JC. Adamantinomas of long bones. Cancer 1974; 34:1796–1805.

    CAS  PubMed  Google Scholar 

  57. Maki M, Saitoh K, Kaneko Y, Fukayama M, Morohoshi T. Expression of cytokeratin1, 5, 14, 19 and transforming growth factors beta 1, beta 2, beta 3 in osteofibrous dysplasia and adamantinoma: a possible association of transforming growth factor beta with basal cell phenotype promotion. Pathol Int 2000; 50:801–807.

    Article  CAS  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Pathology, Long Island Jewish Medical Center, 270-02 76 Avenus, New Hyde Park, NY 10040, USA

    Leonard B. Kahn

  2. Albert Einstein College of Medicine, Bronx, NY 10461, USA

    Leonard B. Kahn

Authors
  1. Leonard B. Kahn
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Leonard B. Kahn.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Kahn, L.B. Adamantinoma, osteofibrous dysplasia and differentiated adamantinoma. Skeletal Radiol 32, 245–258 (2003). https://doi.org/10.1007/s00256-003-0624-2

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00256-003-0624-2

Keywords

Search

Navigation