Denosumab
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Denosumab : a potential new and innovative treatment option for aneurysmal bone cysts. / Lange, Tobias; Stehling, Christoph; Fröhlich, Birgit; Klingenhöfer, Mark; Kunkel, Philip; Schneppenheim, Reinhard; Escherich, Gabriele; Gosheger, Georg; Hardes, Jendrik; Jürgens, Heribert; Schulte, Tobias L.
In: EUR SPINE J, Vol. 22, No. 6, 01.06.2013, p. 1417-22.Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review
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TY - JOUR
T1 - Denosumab
T2 - a potential new and innovative treatment option for aneurysmal bone cysts
AU - Lange, Tobias
AU - Stehling, Christoph
AU - Fröhlich, Birgit
AU - Klingenhöfer, Mark
AU - Kunkel, Philip
AU - Schneppenheim, Reinhard
AU - Escherich, Gabriele
AU - Gosheger, Georg
AU - Hardes, Jendrik
AU - Jürgens, Heribert
AU - Schulte, Tobias L
PY - 2013/6/1
Y1 - 2013/6/1
N2 - 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.
AB - 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.
KW - Antibodies, Monoclonal, Humanized
KW - Bone Cysts, Aneurysmal
KW - Child
KW - Humans
KW - Magnetic Resonance Imaging
KW - Male
KW - Spinal Diseases
U2 - 10.1007/s00586-013-2715-7
DO - 10.1007/s00586-013-2715-7
M3 - SCORING: Journal article
C2 - 23455951
VL - 22
SP - 1417
EP - 1422
JO - EUR SPINE J
JF - EUR SPINE J
SN - 0940-6719
IS - 6
ER -