Gastric gastrointestinal stromal tumor (GIST) is a mesenchymal neoplasm, typically arising from the muscularis propria layer of the stomach wall. Serosa-derived GIST is rarely seen and has not been reported yet. A 49-year-old Chinese female was presented with marked abdominal distension. Ultrasonography revealed a retroperitoneal tumor adjacent to the stomach wall with an intact structure of five echo layers, indicating a non-stomach tumor origin. Preoperative radiological studies suggested tight tumor-stomach adjacency, which was confirmed by intraoperative dissection. Initial frozen section indicated a pathological diagnosis of spindle cell tumor, which turned out to be a gastric GIST originated from the serosa layer of the stomach wall. The current case demonstrates the rare occurrence of serosa-derived GIST. This case also suggests difficulties in preoperative diagnosis of gastric GISTs, especially when uncommon pathological conditions like rare tumor origins were presented.