Giant cell tumor of bone is a locally aggressive primary bone neoplasm affecting patients aged 20–50 years. Neoplastic stromal cells have a phenotype of immature osteoblasts synthesizing various cytokines and molecular factors leading to excessive activation of osteoclastogenesis and increased osteolysis in giant cell tumor of bone. Long-term treatment with denosumab is recommended for unresectable or disseminated disease.A clinical case of long-term treatment with denosumab of initially generalized giant cell tumor of the right ischial bone with metastases to the lungs is presented. Stabilization of the process was first recorded 3 months after the start of therapy and has been maintained to date. The patient was transferred to a maintenance regimen 3 years after the start of treatment. The treatment was not accompanied by adverse events characteristic of bone-modifying agents.Long-term use of denosumab allowed effective control of the tumor process and maintenance of satisfactory quality of life of the patient. Maintenance regimen of denosumab administration was not accompanied by a decrease in treatment effectiveness.
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