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


Authors: Giantini-Larsen, A. M.; Chakravarthy, V. B.; Barzilai, O.; Newman, W. C.; Wexler, L.; Bilsky, M. H.
Review Title: The role of neoadjuvant denosumab in the treatment of aneurysmal bone cysts: A case series and review of the literature
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. © AANS 2022.
Keywords: adolescent; adult; child; clinical article; human tissue; treatment outcome; retrospective studies; case report; postoperative period; neoadjuvant therapy; nuclear magnetic resonance imaging; diagnostic imaging; vascularization; retrospective study; spine; tertiary health care; case study; spine oncology; denosumab; tumor calcinosis; aneurysmal bone cyst; bone cysts, aneurysmal; rankl; humans; human; male; female; article; x-ray computed tomography; devascularization
Journal Title: Journal of Neurosurgery-Pediatrics
Volume: 30
Issue: 6
ISSN: 1933-0707
Publisher: American Association of Neurological Surgeons  
Date Published: 2022-12-01
Start Page: 547
End Page: 554
Language: English
DOI: 10.3171/2022.8.Peds22314
PUBMED: 36282899
PROVIDER: scopus
DOI/URL:
Notes: Article -- Export Date: 3 January 2023 -- Source: Scopus
Altmetric
Citation Impact
BMJ Impact Analytics
MSK Authors
  1. Leonard H Wexler
    193 Wexler
  2. Mark H Bilsky
    321 Bilsky
  3. William Christopher Newman
    27 Newman