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Abstract
BackgroundParosteal osteosarcomas are usually low-grade tumors, however, sometimes they transform to high-grade tumors, which is named dedifferentiation. This phenomenon has been reported in long bones. Recently, we encountered a patient with dedifferentiated parosteal osteosarcoma occurring in the maxilla. Here, we report a first case of dedifferentiated parosteal osteosarcoma of the head and neck region.Case presentationA 45-year-old Japanese woman with a refractory bone lesion in the maxilla presented to our hospital. A biopsy showed atypical spindle cell proliferation involving dedifferentiated high-grade component, which was diagnosed as dedifferentiated parosteal osteosarcoma. Three cycles of neoadjuvant chemotherapy using ifosfamide and pirarubicin were performed followed by sub-total maxillectomy. Histopathological results showed that neoadjuvant chemotherapy was effective for high-grade component. The decision to perform adjuvant chemotherapy (cisplatin and pirarubicin) was made because distant metastasis has been reported, even in cases with dedifferentiated parosteal osteosarcoma in which complete necrosis of high-grade component was achieved due to neoadjuvant chemotherapy. There was no recurrence 15 months after surgery.ConclusionsDedifferentiated parosteal osteosarcoma can occur in the head and neck region. Chemotherapy including anthracycline anticancer agent could be effective for high-grade component of dedifferentiated parosteal osteosarcoma.
Highlights
Parosteal osteosarcomas are usually low-grade tumors, sometimes they transform to highgrade tumors, which is named dedifferentiation
Dedifferentiated parosteal osteosarcoma can occur in the head and neck region
Chemotherapy including anthracycline anticancer agent could be effective for high-grade component of dedifferentiated parosteal osteosarcoma
Summary
Our report suggests that DPOS can arise from the head and neck region and chemotherapy consisting of THP-ADM and IFO could be effective for high-grade components. Administered None high-dose methotrexate (dose range, 9.5–11.4 g/m2), intra-arterial pirarubicin (dose range, 50–80 mg/m2), and dacarbazine (dose range, 400–600 mg/m2). Authors’ contributions HK and HMi drafted and critically reviewed the manuscript. Author details 1Department of Dentistry and Oral Surgery, Division of Oral and Maxillofacial Surgery, Keio University, School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. Author details 1Department of Dentistry and Oral Surgery, Division of Oral and Maxillofacial Surgery, Keio University, School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. 2Department of Neurosurgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. 3Department of Diagnostic Pathology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. 4Department of Pathology and Oncology, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu 807-8555, Japan. 5Department of Plastic and Reconstructive Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan
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