Abstract
Introduction
The anterior elements of the spine, particularly the odontoid processes, are a rare location for osteoblastomas. Pseudomalignant osteoblastomas are themselves rare histologic types and are also extremely rare in this location. Most osteoblastomas are Enneking stage 2 lesions; less frequently, they can be more aggressive with extra-capsular extension (Enneking stage 3). En bloc resection is recommended for aggressive lesions, but the literature is less clear regarding the approach to stage 2 tumors, particularly those with pseudomalignant histologic features.
Case report
A 6-year-old male child presented with a type III pathologic fracture of the odontoid. The fracture healed but upon 6-month follow-up CT scanning, an expansile lesion was detected. Surgical biopsy revealed an osteoblastoma which was treated with intralesional excision. Meanwhile, the excised specimen showed histological features of a pseudomalignant osteoblastoma. Despite this diagnosis, no further treatment was undertaken. At a 10-year follow-up, the patient was free from pain and had full range of motion of the cervical spine; no recurrence was detected.
Conclusion
This unique case of odontoid osteoblastoma illustrates that malignant behavior may not be predicted only by the presence of pseudomalignant features on histology.
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References
Jaffe HL (1956) Benign osteoblastoma. Bull Hosp Joint Dis 17(2):141–151
Lichtenstein L (1956) Benign osteoblastoma: a category of osteoid-and bone-forming tumors other than classical osteoid osteoma, which may be mistaken for giant-cell tumor or osteogenic sarcoma. Cancer 9(5):1044–1052
Janin Y, Epstein JA, Carras R, Khan A (1981) Osteoid osteomas and osteoblastomas of the spine. Neurosurgery 8(1):31–38
Saccomanni B (2009) Osteoid osteoma and osteoblastoma of the spine: a review of the literature. Curr Rev Musculoskelet Med 2(1):65–67
Frassica FJ, Waltrip RL, Sponseller PD, Ma LD, McCarthy EF (1996) Clinicopathologic features and treatment of osteoid osteoma and osteoblastoma in children and adolescents. Orthop Clin North Am 27(3):559–573
Denaro V, Denaro L, Papalia R, Marinozzi A, Di Martino A (2007) Surgical management of cervical spine osteoblastomas. Clin Orthop Relat Res 455:190–195
Raskas DS, Graziano GP, Herzenberg JE, Heidelberger KP, Hensinger RN (1992) Osteoid osteoma and osteoblastoma of the spine. Clin Spine Surg 5(2):204–211
Bruneau M, Cornelius JF, George B (2005) Osteoid osteomas and osteoblastomas of the occipitocervical junction. Spine 30(19):E567–E571
Hladky JP, Lejeune JP, Singer B, Lecomte-Houcke M, Herbaux B, Dhellemmes P (1994) Osteoblastoma of the odontoid process. Pediatr Neurosurg 21(4):260–262
Mori Y, Takayasu M, Saito K, Seki Y, Shibuya M, Yoshida J (1998) Benign osteoblastoma of the odontoid process of the axis: a case report. Surg Neurol 49(3):274–277
Daszkiewicz P, Roszkowski M, Barszcz S, Kuczyński D, Grajkowska W (2004) Osteoblastoma of the dens axis in a 14-year-old boy. A case report. Neurol Neurochir Pol 39(5):420–423
Dahlin DC, Johnson EW (1954) Giant osteoid osteoma. J Bone Joint Surg Am 36(3):559–572
Jaffe HL (1935) Osteoid-osteoma: a benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg 31(5):709–728
Kan P, Schmidt MH (2008) Osteoid osteoma and osteoblastoma of the spine. Neurosurg Clin N Am 19(1):65–70
Marsh BW, Bonfiglio M, Brady LP, Enneking WF (1975) Benign osteoblastoma: range of manifestations. J Bone Joint Surg Am 57(1):1–9
Tani T, Okada K, Shoji K, Hashimoto M, Sageshima M (2000) Osteoblastoma-like osteosarcoma. Skeletal Radiol 29(11):656–659
Mirra JM, Kendrick RA, Kendrick RE (1976) Pseudomalignant osteoblastoma versus arrested osteosarcoma. Cancer 37:2005–2014
Azouz EM, Kozlowski K, Marton D, Sprague P, Zerhouni A, Asselah F (1986) Osteoid osteoma and osteoblastoma of the spine in children. Pediatr Radiol 16(1):25–31
Lichtenstein L (1952) Osteogenic fibroma of bone. Bone Tumors Ed 1:82–87
Lichtenstein L (1956) Benign osteoblastoma. A category of osteoid and bone-forming tumors other than classical osteoid osteoma, which may be mistaken for giant-cell tumor or osteogenic sarcoma. Cancer 9(5):1044–1052
Harrop JS, Schmidt MH, Boriani S, Shaffrey CI (2009) Aggressive “benign” primary spine neoplasms: osteoblastoma, aneurysmal bone cyst, and giant cell tumor. Spine 34(22S):S39–S47
Enneking WF, Spanier SS, Goodman MA (1980) A system for the surgical staging of musculoskeletal sarcoma. Clin Orthop Relat Res 153:106–120
Jawad UM, Scully SP (2010) Classifications in brief: enneking classification: benign and malignant tumors of the musculoskeletal system. Clin Orthop Relat Res 468(7):2000–2002
Cheung FMF, Wu WC, Lam CK, Fu YK (1997) Diagnostic criteria for pseudomalignant osteoblastoma. Histopathology 31(2):196–200
Resnicke D, Kyriakos M, Greenway G (1995) Tumors and tumor-like lesions of bone: imaging and pathology of specific lesions. In: Resnicke D (ed) Diagnosis of bone and joint disorders, 3rd edn. W.B. Saunders Company, Philadelphia, pp 3647–3657
Lucas DR, Unni KK, McLeod RA, O’Connor MI, Sim FH (1994) Osteoblastoma: clinicopathologic study of 306 cases. Hum Pathol 25(2):117–134
Della Rocca C, Huvos AG (1996) Osteoblastoma: varied histological presentations with a benign clinical course: an analysis of 55 cases. Am J Surg Pathol 20(7):841–850
Boriani S, Amendola L, Bandiera S, Simoes CE, Alberghini M, Di Fiore M, Gasbarrini A (2012) Staging and treatment of osteoblastoma in the mobile spine: a review of 51 cases. Eur Spine J 21(10):2003–2010
Flemming DJ, Murphey MD, Carmichael BB, Bernard SA (2000) Primary tumors of the spine. Seminars Musculoskelet Radiol 4(3):299–320
Hosalkar HS, Garg S, Moroz L, Pollock A, Dormans JP (2005) The diagnostic accuracy of MRI versus CT imaging for osteoid osteoma in children. Clin Orthop Relat Res 433:171–177
Davies M, Cassar-Pullicino VN, Davies MA, McCall IW, Tyrrell PN (2002) The diagnostic accuracy of MR imaging in osteoid osteoma. Skelet Radiol 31(10):559–569
Greenspan A, Remagen W (1997) Differential diagnosis of tumors and tumor-like lesions of bones and joints. Lippincott-Raven, Philadelphia
Arkader A, Dormans JP (2008) Osteoblastoma in the skeletally immature. J Pediatric Orthop 28(5):555–560
Dorfman HD, Weiss SW (1984) Borderline osteoblastic tumors: problems in the differential diagnosis of aggressive osteoblastoma and low-grade osteosarcoma. Semin Diagn Pathol. 1(3):215–234
Schajowicz F, Lemos C (1976) Malignant osteoblastoma. Bone Joint J 58(2):202–211
Dehner LP, O’Sullivan MJ, Strauss BL, Wold LE, McAlister WH (2001) Skeletal system: congenital, developmental, and acquired disorders. Pediatric Pathol 2:1361–1362
Bhusnurmath S, Hoch B (2012) Benign bone-forming tumors: approach to diagnosis and current understanding of pathogenesis. Surg Pathol Clin 5(1):101–116
Acknowledgements
Acknowledgements to Professor Afonso Fernandes past head of pathology department of University Hospital of Santa Maria for his critical input in following this patient.
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Fernandes, P., do Brito, J.S., Costa, A. et al. Pseudomalignant osteoblastoma of the odontoid process. Eur Spine J 27 (Suppl 3), 477–482 (2018). https://doi.org/10.1007/s00586-018-5500-9
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DOI: https://doi.org/10.1007/s00586-018-5500-9