Abstract
Simple SummarySpine giant cell tumors (SGCTs) are intermediate malignant bone tumors, sometimes aggressive and responsible for debilitating axial pain and sensorimotor impairments. Non-surgical therapies, including denosumab, radiotherapy, and selective arterial embolization (SAE), have shown promising results in the treatment of patients with inoperable SGCTs. In this systematic review, we aimed to comprehensively analyze the current literature on denosumab, radiotherapy, and SAE for inoperable SGCTs, comparing treatment outcomes and complications using a random-effect model meta-analysis. We found that all three treatments were equally effective in providing symptom improvement and radiological tumor response, also showing low and comparable rates of treatment-related complications. Patients treated with denosumab showed lower rates of local recurrences and distant metastases.Background: Surgical resection remains the preferred treatment in spine giant cell tumors (SGCTs), but it is not always feasible. Conservative strategies have been studied for inoperable cases. We systematically reviewed the literature on inoperable SGCTs treated with denosumab, radiotherapy or selective arterial embolization (SAE). Methods: PubMed, Scopus, Web-of-Science, Ovid-EMBASE, and Cochrane were searched following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines to include studies of inoperable SGCTs treated with denosumab, radiotherapy or SAE. Treatment outcomes were analyzed and compared with a random-effect model meta-analysis. Results: Among the 17 studies included, 128 patients received denosumab, 59 radiotherapy, and 43 SAE. No significant differences in baseline patient characteristics were found between the three groups. All strategies were equally effective in providing symptom improvement (p = 0.187, I2 = 0%) and reduction in tumor volume (p = 0.738, I2 = 56.8%). Rates of treatment-related complications were low (denosumab: 12.5%; radiotherapy: 8.5%; SAE: 18.6%) and comparable (p = 0.311, I2 = 0%). Patients receiving denosumab had significantly lower rates of local tumor recurrence (10.9%) and distant metastases (0%) compared to patients receiving radiotherapy (30.5%; 8.5%) or SAE (35.6%; 7%) (p = 0.003, I2 = 32%; p = 0.002, I2 = 47%). Denosumab was also correlated with significantly higher overall survival rates at 18 months (99.2%) and 24 months (99.2%) compared to radiotherapy (91.5%; 89.6%) and SAE (92.5%; 89.4%) (p = 0.019, I2 = 8%; p = 0.004, I2 = 23%). Mortality was higher in patients receiving SAE (20.9%) or radiotherapy (13.6%) compared to denosumab (0.8%) (p < 0.001), but deaths mostly occurred for unrelated diseases. Conclusions: Denosumab, radiotherapy, and SAE are safe and effective for inoperable SGCTs. Clinical and radiological outcomes are mostly comparable, but denosumab may provide superior tumor control.
Highlights
Spine giant cell tumors (SGCTs) account for approximately 3% of all giant cell tumors of the bones (GCTBs), with a higher incidence in female patients aged 20–40 years [1,2].Recently defined as intermediate malignant bone tumors, they are characterized by a osteolytic and locally aggressive nature, and commonly metastasize to the lungs [3]
Cervical and thoracic spine giant cell tumors (SGCTs) have been reported to be even rarer entities, as we have found among studies
We found that denosumab, locoregional radiotherapy, and selective arterial embolization (SAE) were associated with high rates of symptom improvement and pain relief in patients with inoperable SGCTs, to previous reports on other inoperable GCTBs and/or resected SGCTs [8,10,17]
Summary
Spine giant cell tumors (SGCTs) account for approximately 3% of all giant cell tumors of the bones (GCTBs), with a higher incidence in female patients aged 20–40 years [1,2].Recently defined as intermediate malignant bone tumors, they are characterized by a osteolytic and locally aggressive nature, and commonly metastasize to the lungs [3]. Selective arterial embolization (SAE) proved to be effective in relieving pain, stabilizing lesions, and improving survival by causing tumor shrinkage and/or ossification, owing to the high vascularity of SGCTs [10,11]. We systematically reviewed the literature on inoperable SGCTs treated with denosumab, radiotherapy or selective arterial embolization (SAE). Reviews and Meta-Analyses (PRISMA) guidelines to include studies of inoperable SGCTs treated with denosumab, radiotherapy or SAE. Results: Among the 17 studies included, 128 patients received denosumab, 59 radiotherapy, and 43 SAE. Rates of treatment-related complications were low (denosumab: 12.5%; radiotherapy: 8.5%; SAE: 18.6%) and comparable (p = 0.311, I2 = 0%). Patients receiving denosumab had significantly lower rates of local tumor recurrence (10.9%) and distant metastases (0%) compared to patients receiving radiotherapy (30.5%; 8.5%) or SAE (35.6%; 7%) (p = 0.003, I2 = 32%; p = 0.002, I2 = 47%). Denosumab was correlated with significantly higher overall survival rates at 18 months (99.2%) and 24 months (99.2%)
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