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¹Department of Basic Oncology, Istanbul University, Oncology Institute, Cancer Genetic Division, İstanbul-Turkey DOI : 10.5505/tjo.2019.1975 Osteosarcoma (OS) is the most common bone cancer in children and young adults. Most cases are high grade and aggressive. Studies on the OS genetics have become important in recent years. The reason for the small number of studies on OSs is because the tumor is rarely detected. OSs mostly develop in the femur, tibia, and humerus. Overt metastasis may be detected in 20%?25% of cases. The first genetic studies of OSs were the studies conducted at the cytogenetic level. They showed that OSs had aneuploidic characteristics. Because studies for identifying the gene expression levels used the cytogenetic and molecular analyses, substantial data were available on OSs. In addition, OSs have been found to have a wide miRNA spectrum. The results obtained particularly from the circRNA and miRNA studies suggested that these molecules might be used as biomarkers in the identification of the drug resistance, which reveals the importance of genetic changes in the follow-up of diagnosis and treatment. The aim of the present review was to collect the cytogenetic and molecular studies of OSs and to summarize the genetic data of OSs. Keywords : Cytogenetics; molecular genetics; oncology; osteosarcoma