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Adamantinoma in childhood: report of six cases and review of the literature

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Abstract

Background

Adamantinoma is a rare slow-growing malignant bone tumour.

Objective

To describe the imaging appearances of six childhood cases and review the published literature.

Materials and methods

The database of the Working Group on Paediatric Oncology, Academic Medical Centre/Emma Children’s Hospital Amsterdam, was searched for cases of adamantinoma. Additionally a literature study was performed to identify cases of adamantinoma in childhood.

Results

We identified six local cases of adamantinoma of the long bones, two boys (age 3 and 8 years) and four girls (mean age 8.8 years, range 3.0–14.0 years). The location of the tumour was the tibia in five and the tibia and fibula in one patient. In two patients initially a different diagnosis was made, which led to a delay in appropriate treatment. None of the children showed pulmonary metastases and all underwent total gross resection. On follow-up (mean 6.1 years, range 1.6–12.0 years) all children remained disease-free. Besides a discussion of our six patients, imaging features, histopathology, surgical approach and a literature review of childhood adamantinomas is presented.

Conclusion

Although the incidence of adamantinoma is low, it is important to recognize this rare bone tumour, since in the early stages of the disease adequate treatment will result in an excellent prognosis.

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References

  1. Mirra JM (1989) Adamantinoma and fibrous dysplasia. In: Mirra JM (ed) Bone tumors, 1st edn. Lea & Febiger, Philadelphia, pp 1203–1231

    Google Scholar 

  2. Maier C (1900) Ein primäres myelogenes Plattenepithel-carcinom der Ulna. Bruns’ Beitr Klin Chir 26:533

    Google Scholar 

  3. Fischer B (1913) Uber ein primäres adamantinom des tibia. Z Pathol 12:422–441

    Google Scholar 

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

    PubMed  Google Scholar 

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

    Article  PubMed  CAS  Google Scholar 

  6. Bohndorf K, Nidecker A, Mathias K, et al (1992) The radiological findings in adamantinoma of the long tubular bones (in German). Rofo 157:239–244

    PubMed  CAS  Google Scholar 

  7. Benassi MS, Campanacci L, Gamberi G, et al (1994) Cytokeratin expression and distribution in adamantinoma of the long bones and osteofibrous dysplasia of tibia and fibula. An immunohistochemical study correlated to histogenesis. Histopathology 25:71–76

    Article  PubMed  CAS  Google Scholar 

  8. Drymoussis MD, Schwameis E, Dominikus M, et al (2004) Adamantinoma: a rare primary malignant tumor of long bones. Our clinical experience and a revue of the literature. Acto Orthop Traumatol Hellenica http://www.acta-ortho.gr/v52t1_4.html

  9. Setty NHA, Desai RS (2002) Adamantinoma in the distal end of tibia. Indian J Radiol Imag 12:543–544

    Google Scholar 

  10. Adler CP (1990) Case report 587: adamantinoma of the tibia mimicking osteofibrous dysplasia. Skeletal Radiol 19:55–58

    Article  PubMed  CAS  Google Scholar 

  11. Alguacil-Garcia A, Alonso A, Pettigrew NM, et al (1984) Osteofibrous dysplasia of the tibia and fibula and adamantinoma. Am J Clin Pathol 82:470–474

    PubMed  CAS  Google Scholar 

  12. Altmannsberger M, Poppe H, Schauer A, et al (1982) An unusual case of adamantinoma of long bones. J Cancer Res Clin Oncol 104:315–320

    Article  PubMed  CAS  Google Scholar 

  13. Bertoni F, Zucchi V, Mapelli MD, et al (1988) Case report 506: adamantinoma of the soft tissues of leg. Skeletal Radiol 17:522–526

    Article  PubMed  CAS  Google Scholar 

  14. Biswas SK, Kundu RN, Mukherjee AK, et al (1980) Adamantinoma of tibia. Indian J Pathol Microbiol 23:293–298-B

    PubMed  CAS  Google Scholar 

  15. Bourne MH, Wood MB, Shives TC, et al (1988) Adamantinoma of the radius. A case report. Orthopedics 11:1565–1566

    PubMed  CAS  Google Scholar 

  16. Braud P, Tomeno B, Courpied P, et al (1987) Adamantinoma of the long bones; seven cases treated with en block resection. Fr J Orthop Surg 1:3–12

    Google Scholar 

  17. Campanacci M, Giunti A, Bertoni F, et al (1981) Adamantinoma of the long bones. The experience at the Instituto Ortopedico Rizzoli. Am J Surg Pathol 5:533–542

    Article  PubMed  CAS  Google Scholar 

  18. Changus GW, Speed JS, Steward FW, et al (1957) Malignant angioblastoma of bone. A reappraisal of adamantinoma of long bones. Cancer 10:540–559

    Article  PubMed  CAS  Google Scholar 

  19. Cohn BT, Brahms MA, Froimson AI, et al (1986) Metastasis of adamantinoma sixteen years after knee disarticulation. Report of a case. J Bone Joint Surg Am 68:772–776

    PubMed  CAS  Google Scholar 

  20. Dameron TB (1979) Adamantinoma of the appendicular skeleton. Johns Hopkins Med J 45:107–111

    Google Scholar 

  21. De Keyser F, Vansteenkiste J, Van Den Brande P, et al (1990) Pulmonary metastasis of tibia adamantinoma: case report and review of literature. Acta Clin Belg 45:31–33

    PubMed  Google Scholar 

  22. De Santis E, Porfiri B (1977) L’adamantinoma delle ossa lunghe: aspetti ultrastrutturale di un adamantinoma della tibia. Arch Putti Chir Organi Mov 28:129

    PubMed  Google Scholar 

  23. Durroux R, Ducoin H, Gaubert J, et al (1993) Adamantinoma of the tibia and osteofibrodysplasia. Report of a case. Ann Pathol 13:336–340

    PubMed  CAS  Google Scholar 

  24. Gaubert J, Durroux R (1995) Adamantinoma and osteofibrous dysplasia of the tibia. Rev Chir Orthop Reparatrice Appar Mot 81:333–337

    PubMed  CAS  Google Scholar 

  25. 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 Joint Surg Am 69:1177–1188

    PubMed  CAS  Google Scholar 

  26. Hazelbag HM, Taminiau AH, 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 behavior. J Bone Joint Surg Am 76:1482–1499

    PubMed  CAS  Google Scholar 

  27. 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 Cytogenet 97:5–11

    Article  PubMed  CAS  Google Scholar 

  28. Hebbel R (1940) Adamantinoma of the tibia. Surgery 7:860–868

    Google Scholar 

  29. Henneking K, Rehm KE, Schulz A, et al (1984) Adamantinoma of the long tubular bones. Case report of a fibular tumor. Chirurg 55:407–410

    PubMed  CAS  Google Scholar 

  30. Ishida T, Iijima T, Kikuchi F, et al (1992) A clinicopathological and immunohistochemical study of osteofibrous dysplasia, differentiated adamantinoma and adamantinoma. Skeletal Radiol 21:493–502

    PubMed  CAS  Google Scholar 

  31. Judmaier W, Peer S, Krejzi T, et al (1998) MR findings in tibial adamantinoma. A case report. Acta Radiol 39:276–278

    Article  PubMed  CAS  Google Scholar 

  32. Kanamori M, Antonescu CR, Scott M, et al (2001) Extra copies of chromosomes 7, 8, 12, 19, and 21 are recurrent in adamantinoma. J Mol Diagn 3:16–21

    PubMed  CAS  Google Scholar 

  33. Knapp RH, Wick MR, Scheithauer BW, et al (1982) Adamantinoma of bone. An electron microscopic and immunohistochemical study. Virchows Arch A Pathol Anat Histopathol 398:75–86

    Article  PubMed  CAS  Google Scholar 

  34. Kumar D, Mulligan ME, Levine AM, et al (1998) Classic adamantinoma in a 3-year-old. Skeletal Radiol 27:406–409

    Article  PubMed  CAS  Google Scholar 

  35. Levack B, Revell PA, Roper BA, et al (1986) Adamantinoma associated with fibrous dysplasia. Int Orthop 10:253–259

    Article  PubMed  CAS  Google Scholar 

  36. Madsen MR, Astrup J (1984) Tibial adamantinoma. Ugeskr Laeger 146:2401–2403

    PubMed  CAS  Google Scholar 

  37. Mandahl N, Heim S, Rydholm A, et al (1989) Structural chromosome aberrations in an adamantinoma. Cancer Genet Cytogenet 42:187–190

    Article  PubMed  CAS  Google Scholar 

  38. Mohler DG, Cunningham DC (1997) Adamantinoma arising in the distal fibula treated with distal fibulectomy: a case report and review of the literature. Foot Ankle Int 18:746–751

    PubMed  CAS  Google Scholar 

  39. Moon NF (1965) Adamantinoma of the appendicular skeleton. A statistical review of reported cases and inclusion of 10 new cases. Clin Orthop 43:189–213

    PubMed  CAS  Google Scholar 

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

    PubMed  Google Scholar 

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

    Article  PubMed  CAS  Google Scholar 

  42. Patzak H-J, Kohler V, Henneking K, et al (1990) Ein Adamantinom der Fibula-Verlaufskontrolle. Unfallchirurgie 16:47–49

    Article  PubMed  CAS  Google Scholar 

  43. Perez-Atayde AR, Kozakewich HP, Vawter GF, et al (1985) Adamantinoma of the tibia. An ultrastructural and immunohistochemical study. Cancer 55:1015–1023

    Article  PubMed  CAS  Google Scholar 

  44. Richter CS (1930) Ein fall von adamantinomartiger geschwulst des schienbeins. Z Krebsforsch 32:272–279

    Google Scholar 

  45. Rosen RS, Schwinn CP (1966) Adamantinoma of limb bone: malignant angioblastoma. AJR 97:727–732

    CAS  Google Scholar 

  46. Sarisozen B, Durak K, Ozturk C, et al (2002) Adamantinoma of the tibia in a nine-year-old child. Acta Orthop Belg 68:412–416

    PubMed  CAS  Google Scholar 

  47. Schajowicz F, Santini-Araujo E (1989) Adamantinoma of the tibia masked by fibrous dysplasia. Report of three cases. Clin Orthop 238:294–301

    PubMed  Google Scholar 

  48. Sozzi G, Miozzo M, Di Palma S, et al (1990) Involvement of the region 13q14 in a patient with adamantinoma of the long bones. Hum Genet 85:513–515

    Article  PubMed  CAS  Google Scholar 

  49. Tabei SZ, Abdollahi B, Nilli F, et al (1988) Diagnosis of metastatic adamantinoma of the tibia by pulmonary brushing cytology. Acta Cytol 35:579–581

    Google Scholar 

  50. Ueda Y, Roessner A, Bosse A, et al (1991) Juvenile intracortical adamantinoma of the tibia with predominant osteofibrous dysplasia-like features. Pathol Res Pract 187:1039–1043

    PubMed  CAS  Google Scholar 

  51. Usui K, Yamamoto W, Shuke N, et al (2000) Scintigraphic findings of tibial adamantinoma on a three-phase bone scan. Clin Nucl Med 25:1057–1058

    Article  PubMed  CAS  Google Scholar 

  52. van Haelst UJ, de Haas van Dorsser AH (1975) A perplexing malignant bone tumour. Highly malignant so-called adamantinoma or non-typical Ewing’s sarcoma. Virchows Arch A Pathol Anat Histol 365:63–74

    Article  PubMed  Google Scholar 

  53. Van Schoor JX, Vallaeys JH, Joos GF, et al (1991) Adamantinoma of the tibia with pulmonary metastases and hypercalcemia. Chest 100:279–281

    PubMed  Google Scholar 

  54. Vanhoenacker F, Van Ongeval C, Ceulemans R, et al (1993) Adamantinoma of the tibia: MRI documentation. J Belge Radiol 76:154–156

    PubMed  CAS  Google Scholar 

  55. Vasin VA, Skotnikov VI, Khazov PD, et al (1990) An adamantinoma located in the pelvic bones (in Russian). Vrach Delo 6:96–98

    PubMed  Google Scholar 

  56. Volpe R, Mazabraud A (1984) Adamantinoma of long bones. Pathologica 76:313–322

    PubMed  CAS  Google Scholar 

  57. Zand A, Chamber GH, Street DM, et al (1972). So-called adamantinoma of long bone. Clin Orthop 86:178–182

    Article  PubMed  CAS  Google Scholar 

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

    Article  PubMed  CAS  Google Scholar 

  59. Van der Woude HJ, Hazelbag HM, Bloem JL, et al (2004) MRI of adamantinoma of long bones in correlation with histopathology. AJR 183:1737–1744

    PubMed  Google Scholar 

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Van Rijn, R., Bras, J., Schaap, G. et al. Adamantinoma in childhood: report of six cases and review of the literature. Pediatr Radiol 36, 1068–1074 (2006). https://doi.org/10.1007/s00247-006-0272-5

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