Dedifferentiated parosteal osteosarcoma of the maxilla: a case report and review of the literature

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.

Low‐grade osteosarcoma is predominant in gnathic osteosarcomas: A report of seven cases of osteosarcoma of the jaw

“…Case 4 developed into a high‐grade, non‐osteogenic tumor (dedifferentiation) 4 years after the first presentation to our hospital (Figure 4(a) ), and the details of this case have been described previously (Miyashita et al, 2018 ). Case 7 presented with lung metastasis 2 years after the first presentation, in which histology of the metastasis was composed of a mixture of osteochondrogenic (Figure 4(c) ) and non‐osteogenic (Figure 4(d) ) tumors.…”

“…Furthermore, MDM2 and CDK4 expression were observed on immunostaining in cases 1 (Figure3(a, b)), 4 (Figure3(c,d)), and 7. The histological features of the biopsy specimens are summarized in Tables2 and 3.Case 4 developed into a high-grade, non-osteogenic tumor (dedifferentiation) 4 years after the first presentation to our hospital (Figure4(a)), and the details of this case have been described previously(Miyashita et al, 2018). Case 7 presented with lung metastasis 2 years after the first presentation, in which histology of the metastasis was composed of a mixture of osteochondrogenic (Figure4(c)) and non-osteogenic (Figure 4(d)) tumors.…”

Efficacy of combination-chemotherapy with pirarubicin, ifosfamide, and etoposide for soft tissue sarcoma: a single-institution retrospective analysis

“…However, because most patients with soft tissue sarcoma were pretreated with other chemotherapies, including anthracyclines, the definite evaluation in soft tissue sarcoma was suspended [15]. Since then, there have been various case reports or case series analysis that indicated preferable outcomes with THP-based combination chemotherapy [16][17][18][19]. Thus, a randomized controlled trial for the usage of THP-based chemotherapy will be needed to gain future approval for soft tissue sarcoma.…”

Efficacy of combination-chemotherapy with pirarubicin, ifosfamide, and etoposide for soft tissue sarcoma: A single-institution retrospective analysis

“…However, because most patients with soft tissue sarcoma were pretreated with other chemotherapies, including anthracyclines, the de nite evaluation in soft tissue sarcoma was suspended (14). Since then, there have been various case reports or case series analysis that indicated preferable outcomes with THP-based combination chemotherapy (15,16,17,18). Thus, a randomized controlled trial for the usage of THP-based chemotherapy will be needed to gain future approval for soft tissue sarcoma.…”