Denosumab: a potential new and innovative treatment option for aneurysmal bone cysts

Abstract

Introduction Aneurysmal bone cysts (ABCs) are expansive and destructive lesions positive for osteoclast markers, resembling benign giant cell tumors (GCTs). Treatment options include surgical resection, curettage and cavity filling, embolization, injection of fibrosing agents, or radiotherapy. Particularly in children and adolescents with spinal ABCs, these options may be unsatisfactory, and innovative forms of treatment are needed. Denosumab is a human monoclonal antibody that inhibits osteoclast function by blocking the cytokine receptor activator of the nuclear factor-kappa B ligand. Satisfactory results with denosumab in treating GCTs and immunohistochemical similarities suggest that it may also have positive effects on ABCs. Methods and ResultsThis report is the first description of the therapeutic use of denosumab in two patients with spinal ABCs. Two boys (aged 8 and 11) had recurrent ABCs at C5 after surgery with intralesional tumor resection. Treatment options were discussed by the interdisciplinary tumor board. Arterial embolization was attempted, but failed due to an absence of appropriate afferent arteries. After the families had received extensive information and provided written consent, denosumab therapy was initiated as an individualized treatment, despite the absence as yet of scientific evidence. After the start of denosumab therapy, both patients recovered from pain and neurologic symptoms significantly and are now in a healthy condition with no severe side effects. Magnetic resonance imaging check-ups after 2 or 4 months of denosumab treatment, respectively, showed tumor regression in both patients. Discussion Longer follow-up and clinical studies are warranted to establish the value of denosumab in the treatment of ABCs.

Answer to the Letter to the Editor of H. Namazi concerning “Denosumab: a potential new and innovative treatment option for aneurysmal bone cysts” by Lange T, Stehling C, Fröhlich B, Klingenhöfer M, Kunkel P, Schneppenheim R, Escherich G, Gosheger G, Hardes J, Jürgens H, Schulte TL (2013), Eur Spine J;22(6):1417–1422

“…Our article focused on the clinical application of Denosumab in aneurysmal bone cysts [2]. This comment highlights the physiological mechanisms of the effect of Denosumab and thus perfectly completes and complements our work.…”

Disappearance of giant cells and presence of newly formed bone in the pulmonary metastasis of a sacral giant-cell tumor following denosumab treatment: A case report

“…This inhibits the differentiation of osteoclasts and osteoclast-mediated bone resorption. As the giant cells in GCTB express RANK, denosumab is a promising target-specific therapy for this tumor (14,15). Thomas et al (11) reported the result of an open-label, phase II study with single-agent denosumab in patients who were aged >18 years and had recurrent giant-cell tumors or unresectable tumors.…”

Primary Giant Cell-Rich Lesions of Bone with Role of Denosumab - Histopathological Study with Clinico-Radiological Correlation

“…Pathologist must keep in mind the treatment of the patient while evaluating histopathology of represented material [5][6][7]9,10]. Denosumab has proven to be helpful in treatment of aneurysmal bone cyst [9].…”