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

TL;DR

A patient with a rare jaw cancer achieved long-term remission using a combination of therapies after surgery was not possible.

Contribution

This case report introduces an effective multimodal treatment strategy using Anlotinib for refractory maxillary osteosarcoma.

Findings

The patient achieved complete radiological remission over 48 months with multimodal therapy.

Anlotinib-based treatment was effective in a case where salvage surgery was not feasible.

Multiparametric MRI provided a non-invasive method to confirm recurrence without re-biopsy.

Abstract

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.

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.

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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