PDF(Pubmed)
Abstract：
Giant cell tumor of the bone (GCTB) is a locally aggressive neoplasm where surgery is often curative. However, it can rarely give rise to distant metastases. Currently, the only available active therapeutic option for unresectable GCTB is denosumab, an anti-RANKL monoclonal antibody that dampens the aggressive osteolysis typically seen in this disease. For advanced/metastatic GCTB, denosumab should be continued lifelong, and although it is usually well tolerated, important questions may arise about the long-term safety of this drug. In fact, uncommon but severe toxicities can occur and eventually lead to denosumab discontinuation, such as atypical fracture of the femur (AFF). The optimal management of treatment-related AFF is a matter of debate, and to date, it is unknown whether reintroduction of denosumab at disease progression is a clinically feasible option, as no reports have been provided so far. Hereinafter, we present a case of a patient with metastatic GCTB who suffered from AFF after several years of denosumab; we describe the clinical features, orthopedic treatment, and oncological outcomes, finally providing the first evidence that denosumab rechallenge after AFF occurrence may be a safe and viable option at GCTB progression.
摘要：
骨巨细胞瘤（GCTB）是一种局部侵袭性肿瘤，手术通常可以治愈。然而,它很少会引起远处转移。目前,不可切除的GCTB的唯一有效治疗选择是denosumab，一种抗RANKL单克隆抗体，可以抑制这种疾病中常见的侵袭性骨质溶解。对于晚期/转移性GCTB，denosumab应该终身持续，虽然它通常是很好的耐受性，该药物的长期安全性可能会出现重要问题。事实上,不常见但严重的毒性可能发生，并最终导致denosumab停药，如股骨非典型骨折（AFF）。与治疗相关的AFF的最佳管理是一个有争议的问题，到目前为止，目前尚不清楚在疾病进展时重新引入denosumab是否是临床上可行的选择，因为到目前为止还没有提供任何报告。下文中，我们介绍了一例转移性GCTB患者，该患者经过几年的denosumab治疗后患有AFF；我们描述了临床特征，骨科治疗,和肿瘤的结果，最后提供第一个证据表明,在GCTB进展时,在AFF发生后的denosumab再激发可能是一个安全可行的选择.
