Gastrointestinal stromal tumors (GISTs) are infrequent mesenchymal malignancies arising from the gastrointestinal tract (GIT), accounting for only 0.2% of all GI malignant neoplasms (1). Approximately 60% of GISTs arise in the stomach, 30% in the jejunum and ileum, 4-5% in the duodenum, 4% in the rectum, 1-2% in the colon and appendix, and <1% in the esophagus. Their estimated incidence, including incidental neoplasm, is 10-20 per million (2). The majority are characterized by the oncogenic mutation in either of the two related receptor tyrosine kinases: KIT-CD117 (75-80%) or PDGFRA (platelet-derived growth factor) (5-10%) (3). Recently, extra-gastrointestinal stromal tumors (EGISTs) showing features of GIST have been described at extra-gastrointestinal sites including the omentum, mesentery and retroperitoneal space (4,5). The clinical features and treatment of EGISTs are not well known since there have been only a few cases. To the best of our knowledge, there has been no report of a primary EGIST originating from the broad ligament of the uterus.