Long-term clinical remission in rapidly recurrent maxillary osteosarcoma treated with anlotinib-based multimodal therapy: a case report

安罗替尼联合多模式疗法治疗快速复发性上颌骨骨肉瘤,获得长期临床缓解:病例报告

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Abstract

BACKGROUND: Osteosarcoma of the jaw is a rare clinical entity characterized by a high propensity for local recurrence, for which surgical resection serves as the primary treatment modality. However, achieving negative margins in the maxilla is often complicated by the proximity of critical neurovascular and orbital structures. Furthermore, in certain emergency scenarios, surgical compromises may precipitate suboptimal initial control and rapid disease progression. This presents a significant therapeutic dilemma, particularly given the lack of standardized management guidelines for refractory cases. CASE PRESENTATION: A 37-year-old male underwent emergency orbital decompression for a vision-threatening maxillary mass. One month later, rapid recurrence near the skull base compressed the optic nerve. Due to the risks of hemorrhage and iatrogenic neural injury from re-biopsy, and the patient’s refusal to accept the risk of jeopardizing his preserved vision, a diagnosis of clinicoradiological recurrence was established based on multiparametric MRI (specifically quantitative ADC analysis) and multidisciplinary consensus. The patient received salvage therapy comprising radiotherapy, chemotherapy, and Anlotinib. Over a 48-month follow-up period, the patient achieved complete radiological remission. CONCLUSIONS: While the emergency intervention successfully preserved vision, it also highlights the risks of deviating from standard oncologic principles, which likely precipitated the rapid recurrence. However, the primary significance of this report is to demonstrate that in refractory cases where salvage surgery is contraindicated, an aggressive multimodal strategy—integrating radiotherapy, chemotherapy, and Anlotinib—may serve as a viable salvage option. These findings warrant further validation in prospective trials.

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