A High-grade Sarcoma Arising in a Patient With Recurrent Benign Giant Cell Tumor of the Proximal Tibia While Receiving Treatment With Denosumab
A High-grade Sarcoma Arising in a Patient With Recurrent Benign Giant Cell Tumor of the Proximal Tibia While Receiving Treatment With Denosumab
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DOI：
10.1007/s11999-015-4249-2
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发表时间：
2015-09-01
影响因子：
4.2
通讯作者：
Farfalli, German L.
中科院分区：
文献类型：
--
作者：
Aponte-Tinao, Luis A.;Piuzzi, Nicolas S.;Farfalli, German L.
骨巨细胞瘤是一种原发性良性但局部侵袭性的肿瘤。组织学上典型的骨巨细胞瘤的恶性转化，没有放射治疗，是一种罕见的事件，发生在不到1%的骨巨细胞瘤。虽然手术是标准的初始治疗，但denosumab是一种抑制核因子- κ B配体受体激活剂（RANKL）的单克隆抗体药物，在复发、不可切除和转移性骨巨细胞瘤患者的疾病和症状控制方面显示出相当大的活性。病例描述我们报告一例20岁的女性与复发的良性骨巨细胞瘤，谁有骨肉瘤发展，而接受地诺单抗治疗。文献综述据我们所知，目前还没有低剂量地诺单抗治疗骨质疏松引起感染或恶性肿瘤的报道。然而，虽然报道的副作用相对较少，但用于治疗骨巨细胞瘤的denosumab的安全性和高剂量的不良事件尚未得到很好的定义。Denosumab已成为治疗复发性或不可切除骨巨细胞瘤的重要辅助药物。本例患者在接受denosumab治疗时发生骨巨细胞瘤恶性转化是否由denosumab引起尚不清楚，但如果出现更多病例，应注意这种可能性。未来的研究应侧重于高剂量地诺单抗在不可切除的骨巨细胞瘤患者中的安全性。
Background A giant cell tumor of bone is a primary benign but locally aggressive neoplasm. Malignant transformation in a histologically typical giant cell tumor of bone, without radiotherapy exposure, is an uncommon event, occurring in less than 1% of giant cell tumors of bone. Although surgery is the standard initial treatment, denosumab, a monoclonal antibody drug that inhibits receptor activator of nuclear factor-kappa B ligand (RANKL), has shown considerable activity regarding disease and control of symptoms in patients with recurrence, unresectable, and metastatic giant cell tumors of bone.Case Description We report the case of a 20-year-old woman with a recurrent benign, giant cell tumor of bone, who had a bone sarcoma develop while receiving denosumab treatment.Literature Review To our knowledge, there have been no reports of infection or malignancy with low-dose denosumab administration for osteoporosis. However, while there are relatively few reported side effects, the safety of denosumab and adverse events seen with higher doses, as used in treatment of giant cell tumors of bone are not well defined.Clinical Relevance Denosumab has become a valuable adjunct for treatment of recurrent or unresectable giant cell tumor of bone. It is not clear if our patient's malignant transformation of a giant cell tumor of bone while receiving denosumab treatment was caused by denosumab, but it is important to be aware of the possibility if more cases occur. Future studies should focus on the safety of high-dose denosumab administration in patients with a benign unresectable giant cell tumor of bone.