Malignant transformation of giant cell tumor of bone (GCTB) without radiotherapy exposure is exceptionally rare, occurring in less than 1% of GCTBs. The safety and efficacy of denosumab in patients with GCTB was recently reported. We herein report a case of a benign recurrent GCTB with an H3F3A mutation that underwent secondary malignant transformation during treatment with denosumab. A 29-year-old woman underwent curettage of a GCTB of the left ischium in 2005. Ten years after the first surgery, the GCTB recurred locally. We started treatment with denosumab. During the first 5 months of treatment, we observed a demarcated area of osteosclerosis in the recurrent lesion, and the patient's clinical condition improved. At 6 months, however, the patient developed pain, and a rapidly growing mass was detected by computed tomography. An incisional biopsy was performed. Histologic analysis showed a high-grade osteosarcoma. The patient developed lung metastases and died soon after beginning chemotherapy. The mechanism of sarcomatous transformation of GCTB during denosumab therapy is unclear. These findings suggest that the scientific community should be aware of the possible malignant transformation of GCTB during denosumab treatment.
Download full-text PDF |
Source |
|---|---|
| http://dx.doi.org/10.1093/jjco/hyx112 | DOI Listing |
Publication Analysis
Top Keywords
Similar Publications
Lung transplantation and bone health: A narrative review.
J Heart Lung Transplant
January 2025
Department of Medicine, University Health Network and Sinai Health System, University of Toronto, Toronto, ON, Canada; Toronto General Hospital Research Institute, University Health Network, Toronto, ON, Canada; Joint Department of Medical Imaging, University of Toronto, Toronto, ON, Canada. Electronic address:
Bone health after lung transplantation has not been comprehensively reviewed in over two decades. This narrative review summarizes available literature on bone health in the context of lung transplantation, including epidemiology, presentation and post-operative management. Osteoporosis is reported in approximately 30-50% of lung transplant candidates, largely due to disease-related impact on bone and lifestyle, and corticosteroid-related effects during end-stage lung disease (interstitial lung diseases, chronic obstructive pulmonary disease, and historically cystic fibrosis).
View Article and Find Full Text PDFOsteoporosis in men-East and West: Can the twain meet? A perspective from Asia.
Osteoporos Sarcopenia
December 2024
Osteoporosis and Bone Metabolism Unit, Department of Endocrinology, Singapore General Hospital, Singapore.
Osteoporosis in men remains a significantly underrecognized condition, with notable differences in bone mineral density (BMD) and fracture risk between Asian and Western populations. Despite 30% of hip fractures globally occurring in men, they are less likely to be diagnosed or treated for osteoporosis, especially in resource-limited settings. Given these disparities, a deeper understanding of osteoporosis epidemiology and treatment efficacy in men is essential, particularly in Asian populations.
View Article and Find Full Text PDFChallenges in managing osteogenesis imperfecta in a resource-limited setting: a case report.
J Med Case Rep
January 2025
Lacor Hospital-Gulu, Gulu, Uganda.
Introduction: Osteogenesis imperfecta is a rare inherited connective tissue disorder that results in excessive bone fragility due to defects in collagen production. The majority of osteogenesis imperfecta cases are inherited in an autosomal dominant pattern, and 17 genetic causes have been identified. Diagnosis is usually based on clinical presentation and low bone mineral density scores, while treatment involves a multidisciplinary approach using medical therapies such as bisphosphonates, vitamin C, and pamidronate.
View Article and Find Full Text PDFSuccessful treatment of adult cherubism with a 60 mg denosumab 6-monthly regimen.
JBMR Plus
February 2025
Department of Diabetes and Endocrinology, Royal Melbourne Hospital, Melbourne, VIC 3050, Australia.
Cherubism is a rare autosomal dominant skeletal dysplasia, affecting the maxilla and/or mandible. The condition typically has childhood onset, followed by progression until puberty, with subsequent regression. Cherubism lesions share histological features with giant cell tumor of bone, where high-dose monthly denosumab is an effective medical treatment.
View Article and Find Full Text PDFTranscriptional regulation of Rankl by Txnip-Ecd in aging and diabetic related osteoporosis.
J Adv Res
January 2025
Shanghai Key Laboratory of Orthopedic Implants, Department of Orthopedic Surgery, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200011, China. Electronic address:
Introduction: Bone homeostasis between osteoclast bone resorption and osteoblastic bone formation is tightly regulated by a series of factors such as the receptor activator of nuclear factor-κB ligand (RANKL). Denosumab that neutralizes RANKL is effective and widely applied in the treatment of postmenopausal osteoporosis. However, factors that participated in the RANKL-related bone remodeling process in primary and secondary osteoporosis are less known.
View Article and Find Full Text PDFWant AI Summaries of new PubMed Abstracts delivered to your In-box?
Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!