Secondary osteosarcoma associated with osteofibrous dysplasia: a case report

• Published in: Skeletal radiology 2023-02, Vol.52 (2), p.263-269
• Main Authors: Oka, Naohiro, Hashimoto, Kazuhiko, Nishimura, Shunji, Maenishi, Osamu, Akagi, Masao
• Format: Article
• Language: English
• Subjects: Adolescent; Biopsy; Bone cancer; Bone Diseases, Developmental - pathology; Bone dysplasia; Bone imaging; Bone lesions; Bone Neoplasms - complications; Bone Neoplasms - diagnostic imaging; Cancer; Case Report; Case reports; Chemotherapy; Child, Preschool; Cortical bone; Dysplasia; Fibrous Dysplasia of Bone - complications; Fibrous Dysplasia of Bone - diagnostic imaging; Fractures; Health aspects; Health care facilities; Humans; Imaging; Liquid nitrogen; Magnetic resonance imaging; Male; Malignancy; Medicine; Medicine & Public Health; Neoplasms, Second Primary; Neuroimaging; Nuclear Medicine; Orthopedics; Osteosarcoma; Osteosarcoma - complications; Osteosarcoma - diagnostic imaging; Osteosarcoma - surgery; Pathology; Patients; Radiology; Sarcoma; Soft tissues; Tibia; Tibia - surgery
• ISSN: 0364-2348; 1432-2161
• DOI: 10.1007/s00256-022-04122-5

Secondary osteosarcoma is a rare complication of primary malignancies and benign bone lesions. There are various types of diseases that cause secondary osteosarcoma. A 15-year-old male presented at our medical center complaining of pain and redness in the right lower leg. He had been diagnosed with osteofibrous dysplasia in the right tibia when he was 2 years old and since then had been followed up. Although he had a pathological fracture of the right tibia at the age of 7, his fracture healed with a plaster cast and did not require surgery. At the time of the patient’s last visit, a radiograph revealed a periosteal reaction as well as erosion of the bone cortex. Magnetic resonance imaging revealed an infiltrative area in the soft tissue surrounding the osteofibrous dysplasia lesion in the tibia. Consequent to pathological examination (through bone biopsy), the patient was diagnosed with secondary osteosarcoma. The patient underwent chemotherapy and extensive resection with liquid nitrogen. He has been progressing satisfactorily after the operation. The present case is the first report of secondary osteosarcoma associated with osteofibrous dysplasia. During the long-term follow-up of osteofibrous dysplasia, oncologists should be aware of the possibility of secondary osteosarcoma.