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ICAR SINONASAL TUMORS

TABLE XXIII.B.2 (Continued)

Clinical endpoints

Study

Year LOE Study design Study groups

Conclusion

Mardinger et al. 1266 1. Craniofacial OsS differs from OsS of the long bones in its biological behavior 2. Because of differences in tumor characteristics, chemotherapy did not alter the prognosis 3. 3- and 5-year OS: 90% d and64% d Abbreviations: CSS, chondrosarcoma-specific survival; OS, overall survival; OsS, osteosarcoma; R0, no residual disease; R1, microscopically involved margin; RFS, recurrence-free survival; RT, radiation therapy. a Reported as the ratio between patients affected by a sinonasal osteosarcoma as per the definition reported in Material and Methods and total number of patients; non-osteosarcoma cancers are excluded; number of postirradiation osteosarcomas is reported in round parentheses. b Subsequent row values refer to the entire series and single-patient data could not be extracted. c Extracted from survival curves. d Calculated. 2001 4 Retrospective case series Sinonasal OsS a : 7/13 OS

macroscopic residual disease. In 2.0% patients, the surgical procedure was not described. Chemotherapy and RT were part of the primary treat ment in 48.8% and 24.9% of patients, respectively. Survival outcomes were heterogeneous, with the 3-year OS esti mate ranging from 0% to 100%. In 10 of 19 studies, the 5-year OS estimate was > 50%. Overall, RFS was low, with estimates from 44.9% to 78% at 2–3 years, 40%–60% at 5 years, and 35%–47% at 10 years. Considering the 87 patients with single-patient data available, 1-, 3-, 5-, and 10-year OS estimates were 87.9%, 69.4%, 51.8%, and 47.1%, respectively. Local recurrence was the main modality of treatment failure. Nodal primary involvement and nodal recurrence were rare and limited to high-grade tumors. Distant recurrence was reported in 8.9% of patients. All dis tant metastases occurred in patients with either high-grade osteosarcomas or with no specification on tumor grade. However, cumulative incidence of distant recurrence was 26% and 28% at 5 and 10 years, respectively. 1283 The following negative prognostic factors were iden tified: male gender for RT-associated osteosarcomas, 1270 female gender for primary osteosarcomas, 1275 post-RT presentation, 1283 high/intermediate grade, 1273,1276,1283,1284 nonmaxillary sinonasal/skull base localization, 1273 micro scopic margin involvement, 1273,1276,1277,1279,1280,1283 R2 surgery, 1272 and low-to-null neoplastic bone formation at imaging. 1270 The prognostic role of age is debated. 1276,1282 The prognostic effect of including chemotherapy in treatment, most frequently in the neoadjuvant setting, is controversial: some studies demonstrated a prognostic benefit, 1276,1277 thus advocating for its use in high-grade primary osteosarcomas and RT-associated osteosarcomas, while other studies failed to show a survival advantage. 1280 A single study showed that RT provides a survival bene fit in patients with involved surgical margins or unknown margin status. 1283

Sinonasal osteosarcoma (i.e., including osteosarcoma of the maxilla and adjacent bones) mostly affects patients around their fourth decade of life, with a slight predilec tion for the male gender, and represents roughly 6% of all osteosarcomas. 1285 Secondary osteosarcoma of the sinonasal tract include RT-associated lesions and those arising in the context of Paget’s disease. 1286 Craniofa cial osteosarcoma also represents the most frequent sec ondary tumor in hereditary retinoblastoma survivors pre viously treated with RT. 1287 The maxilla is more frequently involved than other bones of the sinonasal area. 1286 Con ventional osteosarcoma is the most common, with the chondroblastic and osteoblastic subtypes being most fre quently reported. Fibroblastic conventional osteosarcoma has also been reported, whereas nonconventional subtypes such as the telangiectatic, dedifferentiated, and scleros ing are anecdotal. The majority of sinonasal osteosarcomas reported in the literature are of high grade. Most studies on sinonasal osteosarcoma report micro scopic margin status. Approximately half of the patients undergo R0 surgery. Microscopic margin status has been described in several studies as a relevant prognostic fac tor. Other prognostic factors include gender, RT-associated presentation, grade, gross residual disease, and degree of bone formation at imaging. Chemotherapy most likely has a role in the treatment of sinonasal osteosarco mas, although the evidence is not as robust as in pedi atric osteosarcomas. 1288 Roughly half of patients received chemotherapy as part of their primary treatment, most frequently in the neoadjuvant setting. Boon et al. demon strated that neoadjuvant chemotherapy reduces local recurrence in high- and intermediate-grade head and neck osteosarcoma irrespective of age and completeness of resection. 1276 Liao et al. showed increased survival in RT-associated gnathic osteosarcoma treated with surgery and adjuvant chemotherapy over surgery alone. 1277 A