Enteritis cystica profunda (ECP) is a very rare hyperplastic benign lesion of the small intestine. Histologically, it is characterized by mucin-filled cystic spaces frequently lined by benign epithelium within the submucosa and muscularis propria. ECP occurs much less frequently than gastritis cystica profunda (GCP) or colitis cystica profunda (CCP). It is most often seen in adults with Crohn's disease, Peutz-Jeghers syndrome, hamartomatous polyp, or congenital anomalies. We encountered a case of ECP (90 × 15 mm) accompanied by an elongated lipoma in the duodenal bulb, in the absence of any other disease. An endoscopic polypectomy proved sufficient for diagnosing and treating the ECP. To our knowledge, this is the first reported case of ECP accompanied by a lipoma in the duodenal bulb in the absence of any other intestinal disease.

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