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Malignancy in giant cell tumor of bone: analysis of an open-label phase 2 study of denosumab.


ABSTRACT:

Background

Giant cell tumor of bone (GCTB) is a rare osteoclastogenic stromal tumor. GCTB can rarely undergo malignant transformation. This post hoc analysis evaluated and classified malignancies in patients with GCTB who received denosumab.

Methods

This analysis was conducted on patients with pathologically confirmed GCTB and measurable active disease treated with denosumab 120?mg subcutaneously once every 4?weeks, with loading doses on study days 8 and 15, as part of a phase 2, open-label, multicenter study. We identified potential cases of malignancy related to GCTB through an independent multidisciplinary review or medical history, associated imaging or histopathologic reports, and disease course. The findings were summarized and no statistical analysis was performed.

Results

Twenty of five hundred twenty-six patients (3.8%) who received at least one dose of denosumab were misdiagnosed with GCTB that was later discovered to be malignancies: five primary malignant GCTB, five secondary malignant GCTB, four sarcomatous transformations, and six patients with other malignancies (giant cell-rich osteosarcoma, undifferentiated pleomorphic sarcoma, spindle cell sarcoma, osteogenic sarcoma, phosphaturic mesenchymal tumor of mixed connective tissue type, and fibrosarcoma/malignant fibrous histiocytoma). Many malignancies were present before denosumab was initiated (8 definitive cases, 7 likely cases), excluding potential involvement of denosumab in these cases. Signs associated with potential misdiagnoses of GCTB included poor mineralization with denosumab treatment, rapid relapse in pain, or a failure of the typical dramatic improvement in pain normally observed with denosumab.

Conclusions

Although rare, GCTB can undergo malignant transformation, and rates in this study were consistent with previous reports. Signs of poor mineralization or lack of response to denosumab treatment may warrant close monitoring.

Trial registration

clinicaltrials.gov , ( NCT00680992 ). Registered May 20, 2008.

SUBMITTER: Palmerini E 

PROVIDER: S-EPMC7824947 | biostudies-literature | 2021 Jan

REPOSITORIES: biostudies-literature

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Publications

Malignancy in giant cell tumor of bone: analysis of an open-label phase 2 study of denosumab.

Palmerini Emanuela E   Seeger Leanne L LL   Gambarotti Marco M   Righi Alberto A   Reichardt Peter P   Bukata Susan S   Blay Jean-Yves JY   Dai Tian T   Jandial Danielle D   Picci Piero P  

BMC cancer 20210122 1


<h4>Background</h4>Giant cell tumor of bone (GCTB) is a rare osteoclastogenic stromal tumor. GCTB can rarely undergo malignant transformation. This post hoc analysis evaluated and classified malignancies in patients with GCTB who received denosumab.<h4>Methods</h4>This analysis was conducted on patients with pathologically confirmed GCTB and measurable active disease treated with denosumab 120 mg subcutaneously once every 4 weeks, with loading doses on study days 8 and 15, as part of a phase 2,  ...[more]

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