Skip to main content

Advertisement

Log in

Adamantinoma of Long Bones: A Long-term Follow-up Study of 11 Cases

  • Original Paper
  • Published:
Pathology & Oncology Research

Abstract

The aim of this study was to evaluate the clinicopathological features and prognostic significances of 11 histologically proven adamantinoma cases based on an average 12,7 year long follow-up. The male: female ratio was 8:3, aged between 4 and 80 years (mean 29,3 years). The initial diagnosis at referral was other than adamantinoma in six patients (fibrous dysplasia, carcinoma metastasis, osteofibrous dysplasia, bone cyst, non-ossifying fibroma), referring to the differential diagnostic problems. All tumors were localized to the mid part of tibia. By histological evaluation, basaloid pattern on a background of fibrotic stroma dominated in six patients, while spindle and squamous features were less frequently seen. All adamantinomas were positive for cytokeratins often in coexpression with vimentin. No correlation was experienced between histology and clinical outcome. Intralesional curettage (2 pts) was followed by recurrence of the tumor. Wide resection was performed in eight patients with reconstruction using intercalary fibula autografts in seven patients. Reconstruction-related complications occurred in two third of the cases, all of them could however be controlled by repeated surgery. Six recurrences occurred in four patients, two of these recurrences occurred 20 and 16 years after initial surgery. One patient died 9 years after recognition of the tumor of pulmonary metastases. Adamantinoma of the long bones is a low grade malignant tumor, which clinical outcome is difficult to predict based on histology or surgical stage of the tumor. Wide surgical margin, e.g. resection the tumor reduces the rate of recurrence. This study underlines that recurrences do occur even decades after recognition the tumor, therefore a life-long follow-up of the patient is necessary.

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

Access this article

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

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5
Fig. 6
Fig. 7
Fig. 8
Fig. 9

Similar content being viewed by others

References

  1. Fischer B (1913) Über ein primäres Adamantinoma der Tibia. Virchows Arch 12-B:422–441

    Google Scholar 

  2. Dahlin DC, Unni KK (1986) Bone tumors. General aspects and data on 8,542 cases. 4ed Charles C. Thomas, Spingfield

  3. Schajowicz F (1981) Tumors and tumorlike lesions of bone and joints. Springer, New York, Heidelberg, Berlin

    Google Scholar 

  4. Moon MF (1994) Adamantinoma of the appendicular skeleton in children. Int Orthop 18:379–388

    Article  CAS  PubMed  Google Scholar 

  5. Campanacci M (1990) Adamantinoma of the long bones. Bone and Soft Tissue Tumors, 629–638, Springer, New York

  6. Czerniak B, Rojas-Corona RR, Dorfman HD (1989) Morphologic diversity of long bone adamantinoma. The concept of differentiated (regressing) adamantinoma and its relationship to osteofibrous dysplasia. Cancer 64:2319–2334

    Article  CAS  PubMed  Google Scholar 

  7. Huvos AG, Marcove RC (1975) Adamantinoma of long bones. A clinicopathological study of fourteen cases with vascular origin suggested. J Bone and Joint Surg 57-A:148–154

    Google Scholar 

  8. Hazelbag HM, Taminiau AHM, Fleuren GJ et al (1994) Adamantinoma of the long bones. A clinicopathological study of thirty-two patients with emphasis on histological subtype, precursor lesion, and biological behaviour. J Bone and Joint Surg 76-A:1482–1499

    Google Scholar 

  9. Qureshi AA, Shott S, Mallin BA et al (2000) Current trends in the management of adamantinoma of long bones. An international study. J Bone Joint Surg 82-A:1122–1131

    CAS  PubMed  Google Scholar 

  10. Keeney GL, Unni KK, Beabout JW et al (1989) Adamantionoma of long bones. A clinicopathologic study of 85 cases. Cancer 64:730–737

    Article  CAS  PubMed  Google Scholar 

  11. Moon NF, Mori H (1986) Adamantionoma of the appendicular skeleton - updated. Clin Orthop 204:215–237

    PubMed  Google Scholar 

  12. Ulmar B, Delling G, Werner M et al (2006) Classical and atypical location of adamantinomas - presentation of two cases. Onkologie 29:276–278

    Article  PubMed  Google Scholar 

  13. Papagelopoulos PJ, Mavrogenis A, Sim FH et al (2007) Clinicopathological features, diagnosis and treatment of adamantinoma of the long bones. Orthopaedics 30(3):211–215

    Google Scholar 

  14. Huntington TW (1905) Case of bone transference. Use of a segment of fibula to supply a defect in the tibia. Ann Surg 41:249–251

    Article  CAS  PubMed  Google Scholar 

  15. Hicks JD (1954) Synovial sarcoma of the tibia. J Pathol and Bacteriol 67:151–161

    Article  CAS  Google Scholar 

  16. Mori H, Yamamoto S, Hiramatsu K et al (1984) Adamantinoma of the tibia. Ultrastructural and immunohistological study with reference to histogenesis. Clin Orthop 190:299–310

    PubMed  Google Scholar 

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

    Article  CAS  PubMed  Google Scholar 

  18. Hazelbag HM, Wessels JW, Mollevangers P et al (1997) Cytogenetic analysis of adamantinoma of long bones: further indications for a common histogenesis with osteofibrous dysplasia. Cancer Genet and Cytogenet 97:5–11

    Article  CAS  Google Scholar 

  19. Bovee JV, van den Brock LJ, de Boer WI et al (1998) Expression of growth factors and their receptors in adamantionoma of long bones and the implications for its histogenesis. J Pathol 184:24–30

    Article  CAS  PubMed  Google Scholar 

  20. Dorfman HD, Czerniak B (1997) Bone tumors. Mosby, St. Louis

    Google Scholar 

  21. Povysil C, Matejovsky Z (1981) Ultrastructure of adamantinoma of long bones. Virchows Archiv Pathol Anat 393:233–244

    CAS  Google Scholar 

  22. Kahn LB (2003) Adamantinoma, osteofibrous dysplasia and differentiated adamantinoma. Skeletal Radiol 32:245–258

    PubMed  Google Scholar 

  23. Kuruvilla G, Steiner GC (1998) Osteofibrous dysplasia - like adamantinoma of bone: a report of five cases with immunohistochemical and ultrastructural studies. Hum Pathol 29:809–814

    Article  CAS  PubMed  Google Scholar 

  24. Maki M, Athanasou N (2004) Osteofibrous dysplasia and adamantionoma: correlation of proto-oncogene product and matrix protein expression. Hum Pathol 35:69–74

    Article  CAS  PubMed  Google Scholar 

  25. Hatori M, Watanabe M, Hosaka M et al (2006) A classic adamantinoma arising from osteofibrous dysplasia - like adamantionoma in the lower leg: a case report and review of the literature. Tohoku J Exp Med 209:53–59

    Article  PubMed  Google Scholar 

  26. Jundt G, Remberger K, Roessner A et al (1995) Adamantionoma of long bones. A histopathological and immunohistochemical study of 23 cases. Pathol Res Pract 191:112–120

    CAS  PubMed  Google Scholar 

  27. Szendrői M, Rényi-Vámos A, Marschalkó P et al (1994) Csöves csontok adamantinomáinak viselkedése hosszú távú utánkövetés alapján. Magy Traum Ortop 37:37–44

    Google Scholar 

  28. Bély M, Gál D (1982) A case of adamantinoma localized to the tibia. Magyar Traumat 25:75

    Google Scholar 

  29. Bohndorf K, Nidecker A, Mathias K et al (1992) Radiologische Befunde beim Adamantinom der langen Röhrenknochen. Fortschr Roentgenstr 157:239–244

    Article  CAS  Google Scholar 

  30. Van der Woude HJ, Hazelbag HM, Bloem JL, Taminiau AH, Hogendoorn PC (2004) MRI of adamantinoma of long bones in correlation with histopathology. Am J Roentgenol 183:1737–1744

    Google Scholar 

  31. Baker PL, Dockerty MB, Conventry MB (1954) Adamantinoma (so-called) of the long bones. Preview of the literature and a report of three new cases. J Bone and Joint Surg 36-A:704–720

    CAS  Google Scholar 

  32. Gebhardt MC, Lord FC, Rosenberg AE et al (1987) The treatment of adamantinoma of the tibia by wide resection and allograft bone transplantation. J Bone and Joint Surg 69-A:1177–1188

    Google Scholar 

  33. Deijkers RL, Bloem RM, Hogendoorn PC, Verlaan JJ, Kroon HM, Taminiau AH (2002) Hemicortical allograft reconstruction after resection of low-grade malignant bone tumours. J Bone Joint Surg 84-B:1009–1114

    Article  Google Scholar 

  34. Ortiz-Cruz E, Gebhardt MC, Jennings LC et al (1997) The results of transplantation of intercalary allografts after resection of tumors. J Bone and Joint Surg 79-A:97–106

    Google Scholar 

  35. Rock MG, Beabout JW, Unni KK et al (1983) Adamantinoma. Orthopaedics 6:472–477

    Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Miklós Szendrői.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Szendrői, M., Antal, I. & Arató, G. Adamantinoma of Long Bones: A Long-term Follow-up Study of 11 Cases. Pathol. Oncol. Res. 15, 209–216 (2009). https://doi.org/10.1007/s12253-008-9125-x

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12253-008-9125-x

Keywords

Navigation