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

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.

Keywords: RANKL; aneurysmal bone cyst; denosumab; spine oncology.

Publication types

  • Review
  • Research Support, N.I.H., Extramural

MeSH terms

  • Bone Cysts, Aneurysmal* / diagnostic imaging
  • Bone Cysts, Aneurysmal* / drug therapy
  • Bone Cysts, Aneurysmal* / surgery
  • Child
  • Denosumab* / therapeutic use
  • Humans
  • Neoadjuvant Therapy
  • Retrospective Studies
  • Treatment Outcome

Substances

  • Denosumab