Background <p>Osteosarcoma is the most common primary malignant bone tumor, typically affecting adolescents and young adults. While its development is often associated with rapid bone growth and genetic factors, the potential link between metal implants and osteosarcoma remains unclear and rarely reported. This case highlights the unusual occurrence of high-grade osteosarcoma arising at the site of a long-standing metal implant, emphasizing the importance of long-term vigilance in patients with orthopedic hardware.</p> <p>Because implant-associated sarcomas are exceptionally uncommon and causality cannot be inferred from individual cases, detailed clinicopathologic documentation and careful exclusion of alternative etiologies are essential.</p> Case presentation <p>A 49-year-old White middle eastern male presented with progressive pain and swelling in the right thigh. He had sustained an open right femoral shaft fracture in 1990, treated with open intramedullary nailing using a stainless-steel nail with interlocking screws and cerclage wiring. Intermittent symptoms began in 2013, and he was admitted in 2016 with worsening pain and a presumptive diagnosis of chronic osteomyelitis; empiric antibiotics (vancomycin and meropenem) were initiated. Imaging demonstrated an aggressive osteolytic lesion in the distal femoral diaphysis/metadiaphysis adjacent to the prior fixation site, with cortical destruction and an associated soft-tissue component on magnetic resonance imaging (MRI). Core needle biopsy showed chronic inflammation with atypical stromal proliferation, and subsequent open biopsy confirmed high-grade osteogenic osteosarcoma with fibroblastic and chondroblastic differentiation. Microbiological investigations, including blood cultures and multiple intraoperative tissue cultures, were negative, and histopathology did not support osteomyelitis. The patient received perioperative multi-agent chemotherapy with methotrexate, doxorubicin, and cisplatin (MAP) and underwent hip disarticulation with negative margins. No metastatic disease was detected during 9 years of follow-up, and the patient remains disease-free (Table&#xa0;1). This case adds to the limited literature describing implant-associated osteosarcoma and supports ongoing clinical vigilance for late-onset symptoms around orthopedic hardware.</p> Conclusions <p>This case illustrates the diagnostic challenge of distinguishing infection from malignancy in patients with late-onset symptoms around orthopedic hardware. While biologically plausible mechanisms such as chronic local inflammation and metal ion exposure have been proposed, this report should be interpreted as an implant-associated presentation rather than proof of implant-induced carcinogenesis. Persistent or progressive pain or swelling near long-standing implants warrants timely imaging and biopsy.</p>

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Metal implantation and high-grade osteosarcoma of the femur: a case report and review of the literature

  • Mahdi Aghaalikhani,
  • Shobeir Rostami Abusaidi,
  • Farya azimi,
  • Samaneh Aghaalikhani

摘要

Background

Osteosarcoma is the most common primary malignant bone tumor, typically affecting adolescents and young adults. While its development is often associated with rapid bone growth and genetic factors, the potential link between metal implants and osteosarcoma remains unclear and rarely reported. This case highlights the unusual occurrence of high-grade osteosarcoma arising at the site of a long-standing metal implant, emphasizing the importance of long-term vigilance in patients with orthopedic hardware.

Because implant-associated sarcomas are exceptionally uncommon and causality cannot be inferred from individual cases, detailed clinicopathologic documentation and careful exclusion of alternative etiologies are essential.

Case presentation

A 49-year-old White middle eastern male presented with progressive pain and swelling in the right thigh. He had sustained an open right femoral shaft fracture in 1990, treated with open intramedullary nailing using a stainless-steel nail with interlocking screws and cerclage wiring. Intermittent symptoms began in 2013, and he was admitted in 2016 with worsening pain and a presumptive diagnosis of chronic osteomyelitis; empiric antibiotics (vancomycin and meropenem) were initiated. Imaging demonstrated an aggressive osteolytic lesion in the distal femoral diaphysis/metadiaphysis adjacent to the prior fixation site, with cortical destruction and an associated soft-tissue component on magnetic resonance imaging (MRI). Core needle biopsy showed chronic inflammation with atypical stromal proliferation, and subsequent open biopsy confirmed high-grade osteogenic osteosarcoma with fibroblastic and chondroblastic differentiation. Microbiological investigations, including blood cultures and multiple intraoperative tissue cultures, were negative, and histopathology did not support osteomyelitis. The patient received perioperative multi-agent chemotherapy with methotrexate, doxorubicin, and cisplatin (MAP) and underwent hip disarticulation with negative margins. No metastatic disease was detected during 9 years of follow-up, and the patient remains disease-free (Table 1). This case adds to the limited literature describing implant-associated osteosarcoma and supports ongoing clinical vigilance for late-onset symptoms around orthopedic hardware.

Conclusions

This case illustrates the diagnostic challenge of distinguishing infection from malignancy in patients with late-onset symptoms around orthopedic hardware. While biologically plausible mechanisms such as chronic local inflammation and metal ion exposure have been proposed, this report should be interpreted as an implant-associated presentation rather than proof of implant-induced carcinogenesis. Persistent or progressive pain or swelling near long-standing implants warrants timely imaging and biopsy.