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

被引:4
作者
Giantini-Larsen, Alexandra M. [1 ,3 ]
Chakravarthy, Vikram B. [1 ]
Barzilai, Ori [1 ]
Newman, William Christopher [1 ]
Wexler, Leonard [2 ]
Bilsky, Mark H. [1 ,4 ]
机构
[1] Mem Sloan Kettering Canc Ctr, Dept Neurosurg, New York, NY USA
[2] Mem Sloan Kettering Canc Ctr, Dept Pediat, New York, NY USA
[3] Weill Cornell Med Ctr, Dept Neurosurg, New York, NY USA
[4] Mem Sloan Kettering Canc Ctr, New York, NY 10065 USA
基金
美国国家卫生研究院;
关键词
aneurysmal bone cyst; denosumab; spine oncology; RANKL; GIANT-CELL TUMOR; EMBOLIZATION; RANKL; SPINE;
D O I
10.3171/2022.8.PEDS22314
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
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-KB) 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.
引用
收藏
页码:547 / 554
页数:8
相关论文
共 25 条
  • [1] Aneurysmal bone cysts of the spine: treatment options and considerations
    Boriani, Stefano
    Lo, Sheng-fu L.
    Puvanesarajah, Varun
    Fisher, Charles G.
    Varga, Peter P.
    Rhines, Laurence D.
    Germscheid, Niccole M.
    Luzzati, Alessandro
    Chou, Dean
    Reynolds, Jeremy J.
    Williams, Richard P.
    Zadnik, Patti
    Groves, Mari
    Sciubba, Daniel M.
    Bettegowda, Chetan
    Gokaslan, Ziya L.
    [J]. JOURNAL OF NEURO-ONCOLOGY, 2014, 120 (01) : 171 - 178
  • [2] Safety and efficacy of denosumab for adults and skeletally mature adolescents with giant cell tumour of bone: interim analysis of an open-label, parallel-group, phase 2 study
    Chawla, Sant
    Henshaw, Robert
    Seeger, Leanne
    Choy, Edwin
    Blay, Jean-Yves
    Ferrari, Stefano
    Kroep, Judith
    Grimer, Robert
    Reichardt, Peter
    Rutkowski, Piotr
    Schuetze, Scott
    Skubitz, Keith
    Staddon, Arthur
    Thomas, David
    Qian, Yi
    Jacobs, Ira
    [J]. LANCET ONCOLOGY, 2013, 14 (09) : 901 - 908
  • [3] Benign pediatric bone tumors - Evaluation and treatment
    Copley, L
    Dormans, JP
    [J]. PEDIATRIC CLINICS OF NORTH AMERICA, 1996, 43 (04) : 949 - &
  • [4] Serial endovascular embolization as stand-alone treatment of a sacral aneurysmal bone cyst
    Doss, Vinodh T.
    Weaver, Jason
    Didier, Scott
    Arthur, Adam S.
    [J]. JOURNAL OF NEUROSURGERY-SPINE, 2014, 20 (02) : 234 - 238
  • [5] Interest of Denosumab for the Treatment of Giant-cells Tumors and Aneurysmal Bone Cysts of the Spine. About Nine Cases
    Dubory, Arnaud
    Missenard, Gilles
    Domont, Julien
    Court, Charles
    [J]. SPINE, 2016, 41 (11) : E654 - E660
  • [6] Aneurysmal bone cyst: results of an off label treatment with Denosumab
    Duerr, Hans Roland
    Grahneis, Ferdinand
    Baur-Melnyk, Andrea
    Knoesel, Thomas
    Birkenmaier, Christof
    Jansson, Volkmar
    Klein, Alexander
    [J]. BMC MUSCULOSKELETAL DISORDERS, 2019, 20 (01)
  • [7] Dramatic response of aneurysmal bone cyst to denosumab: Case report and literature review
    Fadavi, Pedram
    Arefpour, Amir Mohammad
    Hariri, Ramyar
    Vasheghani, Maryam
    Garousi, Maryam
    Taghizadeh-Hesary, Farzad
    [J]. CLINICAL CASE REPORTS, 2021, 9 (10):
  • [8] Megavoltage radiotherapy for aneurysmal pone cysts
    Feigenberg, SJ
    Marcus, RB
    Zlotecki, RA
    Scarborough, MT
    Berrey, BH
    Enneking, WF
    [J]. INTERNATIONAL JOURNAL OF RADIATION ONCOLOGY BIOLOGY PHYSICS, 2001, 49 (05): : 1243 - 1247
  • [9] Long-term follow-up of Ethibloc injection in aneurysmal bone cysts
    George, Harvey L.
    Unnikrishnan, Puthanveettil Nithin
    Garg, Neeraj K.
    Sampath, Jayanth Sundar
    Bass, Alf
    Bruce, Colin E.
    [J]. JOURNAL OF PEDIATRIC ORTHOPAEDICS-PART B, 2009, 18 (06): : 375 - 380
  • [10] Ghermandi R, 2016, EUR REV MED PHARMACO, V20, P3692