Primary ovarian leiomyosarcomas (POLMs) comprise <0.1% of all ovarian malignancies. Here we aimed to define the clinical, surgical, and pathological features, as well as the oncologic outcome, of POLM. A systematic review of the medical literature was performed to identify articles about POLMs. An electronic literature search was conducted for English language abstracts of articles published between 1975 and December 2018.51 articles were included in the study. The primary endpoint of the study was disease-free survival (DFS) and overall survival (OS), whereas the secondary endpoint was clinicopathological features. Five-year DFS and OS for the entire cohort was 15% and 26%, respectively. The DFS and OS were significantly related to paraaortic lymphadenectomy, a mitotic index >10/high power field, and advanced cancer stages. Eventually, we were unable to obtain clear results, this might be due to the limited number of cases at the literature.With more authors presenting their own cases, it will be possible to have clearer results.