• Qian Chen, Junjie Wu, Kai Zheng, Ming Xu, Ziwei Hou, and Xiuchun Yu.
• First Clinical Medical College, Shandong University of Traditional Chinese Medicine, Jinan, Shandong, China.
• Medicine (Baltimore). 2022 Jul 29; 101 (30): e29812e29812.

RationaleOsteosarcoma (OS) is a primary malignant bone tumor that originates in the mesenchymal tissue. It is the most common type of pleomorphic tumor occurring in children and adolescents. Currently, there is no established systematic treatment for OS that progresses during standard preoperative chemotherapy.Patient Concerns And DiagnosesWe describe a 14-year-old male patient with a 4-month history of pain in the upper right leg. Based on the results of percutaneous biopsy, a diagnosis of OS was made. After admission, the patient was treated with first-line chemotherapy agents. After a single course of treatment, the tumor progressed locally and no limb salvage was feasible.Interventions And OutcomesIntervention with denosumab combined with chemotherapy led to a significant reduction in tumor volume and ossification of soft tissue, which successfully resulted in limb salvage rather than amputation. The patient showed no evidence of recurrent or distant metastasis at 6-month follow-up.LessonsTreatment with receptor activator of nuclear factor-ĸB ligand inhibitor denosumab combined with standard chemotherapy is effective for advanced OS progressing after chemotherapy. We recommend denosumab therapy for successful limb salvage in patients with high-grade OS associated with osteolytic bone destruction and refractory to preoperative neoadjuvant chemotherapy.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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