Atypical (bizarre) leiomyoma is a benign uterine smooth-muscle tumor characterized by a) a significant number of cells with dense eosinophilic cytoplasm and enlarged, bizarre single/multiple hyperchromatic or multiple nuclei without tumor necrosis and b) poor mitotic activity. We report the case of an atypical (bizarre) leiomyoma revealed by focal fluoro-deoxyglucose (FDG) uptake during a PET-CT in a patient with relapsing abdominal and retroperitoneal leiomyosarcoma.