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

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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 Results

This 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.

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Acknowledgments

The authors especially thank Sebastian Retzlaff, MD, for contributing the MRI of patient no. 2.

Conflict of interest

None.

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Authors and Affiliations

  1. Department of Orthopedics and Tumor Orthopedics, Münster University Hospital, Albert-Schweitzer-Campus 1, 48149, Münster, Germany

    Tobias Lange, Georg Gosheger, Jendrik Hardes & Tobias L. Schulte

  2. Department of Clinical Radiology, Münster University Hospital, Münster, Germany

    Christoph Stehling

  3. Department of Pediatric Hematology and Oncology, Münster University Hospital, Münster, Germany

    Birgit Fröhlich & Heribert Jürgens

  4. Department of Neurosurgery, Münster University Hospital, Münster, Germany

    Mark Klingenhöfer

  5. Department of Pediatric Neurosurgery, Altona Children’s Hospital, Hamburg, Germany

    Philip Kunkel

  6. Department of Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany

    Reinhard Schneppenheim & Gabriele Escherich

Authors
  1. Tobias Lange
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  2. Christoph Stehling
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  3. Birgit Fröhlich
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  7. Gabriele Escherich
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  8. Georg Gosheger
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  9. Jendrik Hardes
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  10. Heribert Jürgens
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  11. Tobias L. Schulte
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Correspondence to Tobias Lange.

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Lange, T., Stehling, C., Fröhlich, B. et al. Denosumab: a potential new and innovative treatment option for aneurysmal bone cysts. Eur Spine J 22, 1417–1422 (2013). https://doi.org/10.1007/s00586-013-2715-7

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  • DOI: https://doi.org/10.1007/s00586-013-2715-7

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