Introduction: Colitis Cystica Profunda (CCP) is a rare benign disease of the colon and rectum of uncertain etiology. Its recognition is important since it often mimics malignant processes. We review a case of CCP in a 50-year-old patient, with emphasis on endoscopic ultrasound findings and present review of the current literature. Case description: 50-year-old male with no past medical history was referred to our institution for further evaluation of a large multilobulated mass in the left proximal colon found on screening colonoscopy. We performed colonoscopy with endoscopic ultrasound and fine needle aspiration and snare polypectomy. Colonoscopy up to the transverse colon revealed a large multilobulated submucosal mass with normal overlying mucosa that extends from 50 cm to 70 cm from the anal verge (fig.1). The endosonographic examination revealed anechoic, round, well demarcated lesions with a hyperechoic rim and hypoechoic foci within (fig.2). They were located within the deep mucosa and submucosal layer. The muscularis propria was intact. The largest submucosal lesion measured 18.6 mm x11.4 mm. Small amount of fluid was aspirated by FNA from two lesions. Snare polypectomy of one of the lesions was then performed. The polypectomy site was closed with two endoclips. Histological examination showed multinucleate giant cells in a background of benign colonic elements and mucin consistent with a diagnosis of CCP. Discussion: CCP is an uncommon benign entity that mimics malignancy on endoscopic views but histologically characterized by mucin-filled cysts beneath the muscularis mucosa. It can present in a localized form with a polypoid lesion or more diffusely along the gastrointestinal tract. Endoscopically, CCP are polypoid lesions covered by normal, edematous or ulcerated mucosa. There are few case reports of the endosonographic features of CCP. They were described as hypoechoic or anechoic lesions involving the mucosa or submucosa, with areas of echorefringent fibrosis between lesions by Hulsmans, F.J., et al. In another case report by Sultan et al. the EUS findings of CCP were described as hypoechoic lesion with cystic/spongy features involving the mucosa and submucosa. Our case depicted similar features of well demarcated mucosal and submucosal anechoic lesions with hypoechoic foci surrounded by hyperechoic rim. Colonic EUS can be a useful tool to assist in establishing the diagnosis of CCP.Figure: Colonoscopy showing large multilobulated submucosal mass with overlying normal looking mucosa.Figure: Endosonographic imaging showing anechoic, round, well demarcated lesions.