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新辅助地舒单抗治疗骨巨细胞瘤的作用:病例系列和文献复习。

The role of neoadjuvant denosumab in the treatment of aneurysmal bone cysts: a case series and review of the literature.

机构信息

Departments of1Neurosurgery and.

2Department of Neurosurgery, Weill Cornell Medical Center, New York, New York.

出版信息

J Neurosurg Pediatr. 2022 Sep 30;30(6):547-554. doi: 10.3171/2022.8.PEDS22314. Print 2022 Dec 1.

Abstract

OBJECTIVE

Aneurysmal bone cysts (ABCs) are benign cystic lesions most commonly occurring in the long bones of pediatric patients. Spinal ABCs may be difficult to resect given their invasive, locally destructive nature, proximity to critical structures such as the spinal cord, and their intrinsic hypervascularity, for which complete embolization is often constrained by radiculomedullary segmental feeders. Denosumab, a monoclonal antibody that binds the receptor activator of nuclear factor kappa B (NF-κB) ligand, has been utilized in the treatment of ABCs most often as a rescue therapy for recurrent disease. Here, the authors present 3 cases of neoadjuvant denosumab use in surgically unresectable tumors to calcify and devascularize the lesions, allowing for safer, more complete resection.

METHODS

This is a single-center, retrospective case series treated at a tertiary care cancer center. The authors present 3 cases of spinal ABC treated with neoadjuvant denosumab.

RESULTS

All 3 patients experienced calcification, size reduction, and a significant decrease in the vascularity of their ABCs on denosumab therapy. None of the patients developed new neurological deficits while on denosumab. Subsequently, all underwent resection. One patient continued denosumab during the immediate postoperative period because a subtotal resection had been performed, with stabilization of the residual disease. No complications were associated with denosumab administration.

CONCLUSIONS

The use of denosumab in unresectable ABCs can cause calcification and devascularization, making safe resection more likely.

摘要

目的

动脉瘤样骨囊肿(ABC)是一种良性囊性病变,最常发生于儿童长骨。由于其侵袭性、局部破坏性、靠近脊髓等关键结构以及内在的高血管性,脊柱 ABC 可能难以切除,因此完全栓塞往往受到神经根髓节段供血的限制。地舒单抗是一种单克隆抗体,可结合核因子 kappa B(NF-κB)配体的受体激活剂,已被用于 ABC 的治疗,最常用于复发病例的挽救治疗。在此,作者介绍了 3 例手术不可切除肿瘤中使用地舒单抗的新辅助治疗病例,使病变钙化和血管化,从而更安全、更完全地切除。

方法

这是一项在三级癌症治疗中心进行的单中心回顾性病例系列研究。作者介绍了 3 例脊柱 ABC 患者接受新辅助地舒单抗治疗的情况。

结果

所有 3 例患者在接受地舒单抗治疗后均出现了 ABC 的钙化、体积缩小和血管密度显著降低。在接受地舒单抗治疗期间,没有患者出现新的神经功能缺损。随后,所有患者均接受了手术切除。1 例患者因行不完全切除术且残留疾病稳定而在术后立即接受地舒单抗治疗。地舒单抗治疗无相关并发症。

结论

地舒单抗在不可切除的 ABC 中应用可引起钙化和血管化,从而使安全切除更有可能。

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