Clinical and pathological results of denosumab treatment for giant cell tumors of bone: Prospective study of 14 cases

Abstract Background Pharmacologic options for treatment of osteolytic diseases especially in children are limited. Although not licensed for use, denosumab, a fully humanized antibody to RANKL, is used in children with good effects. Among others, one possible indication are giant cell tumors and aneurysmatic bone cysts. However, there are reports of severe hypercalcemia during weeks to months after termination of denosumab, that are rarely seen in adults. Methods We collected data of four patients, aged 6–17 years, who experienced severe hypercalcemia after completion of treatment with denosumab for unresectable giant cell tumors of bone or aneurysmal bone cysts and methods of their treatment. The detailed case information were described. Results One patient was treated with long-term, high-dose steroid therapy, leading to typical Cushing’s syndrome. Another patient was restarted on denosumab repeatedly due to relapses of hypercalcemia after every stop. Finally, in two patients, hypercalcemia ceased definitely after treatment with bisphosphonates. However, several applications were necessary to stabilize calcium levels. Conclusions There is a considerable risk of hypercalcemia as an adverse effect after denosumab treatment in children. Therapeutic and, preferably, preventive strategies are needed. Bisphosphonates seem to be an option for both, but effective proceedings still remain to be established.

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Faculty Opinions recommendation of Role of the VEGF-Flt-1-FAK pathway in the pathogenesis of osteoclastic bone destruction of giant cell tumors of bone.

Faculty Opinions – Post-Publication Peer Review of the Biomedical Literature ◽

10.3410/f.7679956.7994054 ◽

2011 ◽

Author(s):

Maria Luisa Brandi ◽

Gemma Marcucci

Keyword(s):

Giant Cell ◽

Bone Destruction ◽

Giant Cell Tumors ◽

Tumors Of Bone

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Giant Cell Tumor of the Talar Neck

Journal of the American Podiatric Medical Association ◽

10.7547/0970225 ◽

2007 ◽

Vol 97 (3) ◽

pp. 225-228 ◽

Cited By ~ 7

Author(s):

Hakan Selek ◽

Hamza Özer ◽

Sacit Turanli ◽

Özlem Erdem

Keyword(s):

Giant Cell Tumor ◽

Giant Cell ◽

Clinical Signs ◽

Cell Tumor ◽

Giant Cell Tumors ◽

Radiographic Appearance ◽

Aneurysmal Bone Cysts ◽

Tumors Of Bone ◽

Brown Tumors ◽

Small Bones

We describe a patient with a giant cell tumor in the talar head and neck of the left foot who was diagnosed as having osteochondritis dissecans and treated with arthroscopic drilling in this same location 3 years earlier. Giant cell tumors can be confused with several conditions, including giant cell reparative granulomas, brown tumors, and aneurysmal bone cysts. Giant cell tumors of bone typically occur in the epiphysis of long bones, including the distal femur and proximal tibia. They are uncommonly found in the small bones of the foot or ankle, and talar involvement is rare. Despite this rarity, the radiographic appearance and clinical signs of talar lesions should be considered in the differential diagnosis of nontraumatic conditions in the foot. (J Am Podiatr Med Assoc 97(3): 225–228, 2007)

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Protein Expression Profiling of Giant Cell Tumors of Bone Treated with Denosumab

PLoS ONE ◽

10.1371/journal.pone.0148401 ◽

2016 ◽

Vol 11 (2) ◽

pp. e0148401 ◽

Cited By ~ 11

Author(s):

Kenta Mukaihara ◽

Yoshiyuki Suehara ◽

Shinji Kohsaka ◽

Keisuke Akaike ◽

Yu Tanabe ◽

...

Keyword(s):

Protein Expression ◽

Giant Cell ◽

Expression Profiling ◽

Giant Cell Tumors ◽

Tumors Of Bone

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Giant-Cell Tumors of Bone and Progesterone Receptors

Orthopedics ◽

10.3928/0147-7447-20031201-10 ◽

2003 ◽

Vol 26 (12) ◽

pp. 1209-1212

Author(s):

Nikolaos Demertzis ◽

Fani Kotsiandri ◽

Ioulia Giotis ◽

Nikiphoros Apostolikas

Keyword(s):

Giant Cell ◽

Progesterone Receptors ◽

Giant Cell Tumors ◽

Tumors Of Bone

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A single-center experience with giant cell tumors of sphenoid bone and clivus

Tumori Journal ◽

10.1177/03008916211024357 ◽

2021 ◽

pp. 030089162110243

Author(s):

Büşra Yaprak Bayrak ◽

Emre Özcan ◽

Çiğdem Vural ◽

Atakan Emengen ◽

Burak Çabuk ◽

...

Keyword(s):

Giant Cell ◽

Treatment Algorithm ◽

Residual Tumor ◽

Sphenoid Bone ◽

Giant Cell Tumors ◽

Single Center Experience ◽

Histopathologic Diagnosis ◽

Choice Of Treatment ◽

Transnasal Surgery ◽

Denosumab Treatment

Objective: To present pathologic, clinical, and treatment findings for giant cell tumors (GCTs) of sphenoid bone and clivus. Methods: We describe the optimal treatment algorithm in patients with a histopathologic diagnosis of bone GCT by presenting the effects of denosumab treatment in both pediatric and adult patients with GCT undergoing endoscopic transnasal surgery. Clinicopathologic correlation is crucial for the differential diagnosis of GCT and the choice of treatment modality. Conclusion: GCT of bone is a local aggressive tumor that accounts for about 3%–7% of all bone tumors. GCTs located in the cranium are extremely uncommon neoplasms. There are no defined guidelines for the treatment of GCTs in skull base. Following surgical resection of the tumor, the addition of denosumab treatments to radiotherapy has a significant role in preventing the recurrence of GCT and in promoting regression of residual tumor size.

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