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Abstract

Kemik tümörleri içerisinde nadir görülen osteofibrözdisplazi benzeri adamantinoma (OFDBA)’nın saptandığı ve literatürde bildirilen en küçük yaştaki olgu tanımlanmıştır. OFDBA, osteofibröz displa- zi ve klasik adamantinomaya radyolojik, histopatolojik olarak benzerlik göstermektedir. OFDBA’nın klasik adamantinomaya dönüşebilme özelliği nedeniyle selim bir lezyon kabul edilmesine rağmen, pre- malign bir lezyon olarak değerlendirilmesi önerilir. Olgumuzda da biopsi ile OFDBA tanısı sonrasında lokal rezeksiyon ve masif allogreft ile rekonstrüksiyon uygulanmıştır

Keywords

• Adamantinoma; osteofibröz displazi; osteofibröz displazi benzeri adamantinoma

Osteofibrous dysplasia-like adamantinoma in a 3-month-old male infant: a case report

Abstract

We report a case of a very rarely seen osteofibrous dysplasia-like adamantinoma (OFDLA) of the lower leg in a 3-month-old male infant, making it the youngest case in the literature. OFDLA is typically regarded as a benign lesion; however, due to its convertibility into classical adamantinoma, it is recommended to evaluate it as a pre-malignant lesion. After OFDLA diagnosis with biopsy, our case underwent surgical resection and reconstruction with a large allograft. Patient experienced good outcomes and did not experience any local relapse in the 3-year follow-up.

Keywords

• Adamantinoma
• osteofibrous dysplasia
• osteofibrous dysplasia-like adamantinoma

References

1. Fletcher CDM, Unni KK, Mertens F. Pathology and Ge- netics of Tumors of Soft Tissue and Bone. 1. Ed. Interna- tional Agency for Research on Cancer Press, Lyon, 2002. p. 332–44.
2. Hatori M, Watanabe M, Hosaka M, Sasano H, Narita M, Kokubun S. A classic adamantinoma arising from osteofi- brous dysplasia-like adamantinoma in the lower leg: a case report and review of the literature. Tohoku J Exp Med 2006;209:53–9.
3. Hahn SB, Kim SH, Cho NH, Choi CJ, Kim BS, Kang HJ. Treatment of osteofibrous dysplasia and associated le- sions. Yonsei Med J 2007;48:502–10.
4. Unni KK, Dahlin DC, Beabout JW, Ivins JC. Adamanti- nomas of long bones. Cancer. 1974;34:1796–805.
5. Hazelbag HM, Taminiau AH, Fleuren GJ, Hogendoorn PC. Adamantinoma of the long bones. A clinicopathologi- cal study of thirty-two patients with emphasis on histo- logical subtype, precursor lesion, and biological behavior. J Bone Joint Surg Am 1994;76:1482–99.
6. Yıldız M, Yıldız K, Turhan AU, Baki C, Şener M. A case of adamantinama of the fibu/a. Acta Orthop. Traumatol Turc 1991;25:243–6.
7. Sarisozen B, Durak K, Ozturk C. Adamantinoma of the tibia in a nine-year-old child. Acta Orthop Belg 2002;68:412–6.
8. Pieterse AS, Smith PS, McClure J. Adamantinoma of long bones: clinical, pathological and ultrastructural features. J Clin Pathol 1982;35:780–6.

9. Baker Pl, Dockerty Mb, Coventry Mb. Adamantinoma (so-called) of the long bones; review of the literature and report of three new cases. J Bone Joint Surg Am 1954;36:704–20.
10. Qureshi AA, Shott S, Mallin BA, Gitelis S. Current trends in the management of adamantinoma of long bones. An in- ternational study. J Bone Joint Surg Am 2000;82:1122–31.
11. Rankin JO. Adamantinoma of the tibia. J.Bone Joint Surg. Am. 1939;2:425–32.
12. Khanna M, Delaney D, Tirabosco R, Saifuddin A. Os- teofibrous dysplasia, osteofibrous dysplasia-like adaman- tinoma and adamantinoma: correlation of radiological imaging features with surgical histology and assessment of the use of radiology in contributing to needle biopsy diagnosis. Skeletal Radiol 2008;37:1077–84.
13. Mathew M, Joseph B. Differentiated adamantinoma: a case report and review of literature. Indian J Pathol Mi- crobiol 2007;50:565–7.
14. Kahn LB. Adamantinoma, osteofibrous dysplasia and dif- ferentiated adamantinoma. Skeletal Radiol 2003;32:245– 58.
15. Gebhardt MC, Lord FC, Rosenberg AE, Mankin HJ. The treatment of adamantinoma of the tibia by wide resection and allograft bone transplantation. J Bone Joint Surg Am 1987;69:1177–88.
16. Campanacci M, Laus M. Osteofibrous dysplasia of the tibia and fibula. J Bone Joint Surg Am 1981;63:367–75.
17. Kuruvilla G, Steiner GC. Osteofibrous dysplasia-like adamantinoma of bone: a report of five cases with immu- nohistochemical and ultrastructural studies. Hum Pathol 1998;29:809–14.
18. Acta Orthop Traumatol Turc 212