Pancreatic cysts include a variety of benign, premalignant, and malignant lesions. Endometrial cysts in the pancreas are extremely rare lesions that are difficult to diagnose before surgery. We report the case of a 26-year-old patient with a recent episode of left abdominal pain who presented with a large cyst in the pancreatic body. Laboratory results showed white blood cell and C-reactive protein elevation, whereas the patient's tumor marker levels were within the normal range. Distal pancreatectomy with splenectomy was performed. The final histopathological examination confirmed the presence of endometriotic cysts within the pancreas. Only 13 cases of endometriotic cysts of the pancreas have been previously reported. The preoperative diagnosis is challenging, and most patients undergo pancreatic resection because of suspected neoplasms. This case report reviews previous studies and discusses the clinicopathological features, pathogenesis, and appropriate treatment for pancreatic endometrial cysts.

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