Abstract
Giant cell tumor of bone (GCTB) is a biologically benign and locally aggressive tumor that most often affects the epiphyseal and metaphyseal sites of long bones in the young adult population. Overexpression of receptor activator of nuclear factor kappa B ligand (RANKL) by cancerous mesenchymal stromal cells stimulates a signal transduction cascade that recruits and activates multinucleated osteoclast-like giant cells, resulting in pathologic bone resorption. Denosumab, an RANKL inhibitor that blocks the RANKL-mediated osteoclast activation, has been recently approved by the United States Food and Drug Administration (FDA) for the treatment of aggressive GCTB. Although uncommon, several studies reported drug-related malignant morphological transformation of benign GCTB following treatment with denosumab therapy. The aim of the article was to review the clinicopathological characteristics of all the reported cases of malignant sarcomatous transformation of GCTB after treatment with denosumab therapy in patients without any history of prior exposure to radiotherapy.
Highlights
BackgroundGiant cell tumor of bone (GCTB) is a benign osteolytic neoplasm that primarily involves the epiphyseal and metaphyseal regions of long bones [1]
The patient developed a high-grade spindle cell osteosarcoma in the initial site while receiving preoperative denosumab therapy
From a technical point of view, a potential hypothesis has been proposed to account for the sarcomatous transformation of GCTB that takes place during or after denosumab therapy
Summary
Giant cell tumor of bone (GCTB) is a benign osteolytic neoplasm that primarily involves the epiphyseal and metaphyseal regions of long bones [1]. How to cite this article Alaqaili S I, Abduljabbar A M, Altaho A J, et al (December 28, 2018) Malignant Sarcomatous Transformation of Benign Giant Cell Tumor of Bone after Treatment with Denosumab Therapy: A Literature Review of Reported Cases. The patient developed a high-grade spindle cell osteosarcoma in the initial site (ischium) while receiving preoperative denosumab therapy. From a technical point of view, a potential hypothesis has been proposed to account for the sarcomatous transformation of GCTB that takes place during or after denosumab therapy This hypothesis takes into account that osteosarcoma arising in a histologically benign GCTB may encompass some scattered foci of malignant tumor. It should be noted that confounding factors (for example, age, gender, and previous treatment with selective intra-arterial embolization) may play contributing roles to the malignant sarcomatous transformation seen in GCTB patients treated with denosumab therapy.
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