Abstract
We report the peculiar case of a parosteal osteosarcoma arising beneath the periosteum in a 12-year-old boy. He complained of difficulty in left knee flexion. Plain radiography showed a uniformly dense mineralized mass in the bone cortex and parosteal ossified nodules at the metaphysis and diaphysis of the left distal femur. Periosteal reaction was not evident. Uniquely, plain radiography had a smooth outline and revealed gradually thickening mass toward the center. Histologically, the tumor showed a proliferation of spindle-shaped cells with parallel-oriented dense bone trabeculae and hyaline cartilaginous tissue disclosing mild atypia. The periosteum was inverted along the polypoid mass, but there was no periosteum at the top. Immunohistochemically, the spindle cells, including those at the top of the polypoid mass, and cartilaginous cells were positive for MDM2 and CDK4. MDM2 gene amplification was detected in these cells by fluorescence in situ hybridization. Despite the peculiar feature of plain radiography, the lesion was diagnosed as parosteal osteosarcoma. This case report presents a case of parosteal osteosarcoma arising beneath the periosteum, although it is postulated to arise in the outer layer of the periosteum. The unique radiographic findings in this case suggest an association of parosteal osteosarcoma with vigorous bone growth before closure of the growth plate.
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References
Czerniak B. Dorfman and Czerniak’s bone tumors: Elsevier; 2015. p. 309–28.
Okada K, Frassica FJ, Sim FH, Beabout JW, Bond JR, Unni KK. Parosteal osteosarcoma. A clinicopathological study. J Bone Joint Surg Am. 1994;76(3):366–78.
Schajowicz F, McGuire MH, Santini Araujo E, Muscolo DL, Gitelis S. Osteosarcomas arising on the surfaces of long bones. J Bone Joint Surg Am. 1988;70(4):555–64.
Wang J, Nord KH, O’Donnell PG, Yshida A. Parosteal osteosarcoma: WHO classification of tumors. 5th ed. Soft tissue and bone tumors. Lyon: IARC Press; 2020. p. 410–3.
Wunder JS, Eppert K, Burrow SR, Gokgoz N, Bell RS, Andrulis IL. Co-amplification and overexpression of CDK4, SAS and MDM2 occurs frequently in human parosteal osteosarcomas. Oncogene. 1999;18(3):783–8.
Righi A, Gambarotti M, Benini S, Gamberi G, Cocchi S, Picci P, et al. MDM2 and CDK4 expression in periosteal osteosarcoma. Hum Pathol. 2015;46(4):549–53.
Nodomi S, Umeda K, Okamoto T, Saida S, Hiramatsu H, Watanabe K, et al. A pediatric case of metastatic conventional parosteal osteosarcoma treated with multidrug chemotherapy. Pediatr Blood Cancer. 2016;63(8):1471–3.
Acknowledgements
We greatly appreciate Koji Sagiyama, a skeletal radiologist. He reviewed this case with us and assisted to describe radiological findings.
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All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. This study was approved by the Ethics Committee of our University (nos. 29-429, 29-625).
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Ishihara, S., Kohashi, K., Kuboyama, Y. et al. Parosteal osteosarcoma with a manifestation of subperiosteal low-grade central osteosarcoma. Skeletal Radiol 50, 1903–1907 (2021). https://doi.org/10.1007/s00256-021-03747-2
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DOI: https://doi.org/10.1007/s00256-021-03747-2