Colitis Cystica Profunda (CCP) is an uncommon and benign disease entity characterized by mucoid cysts located in the submucosal layer of the colon. It can mimic cystic submucosal tumors or mucinous adenocarcinoma and is usually located in the rectum. CCP is found in the ascending colon less frequently. CCP manifesting as multiple cystic tumors, similar to pneumatosis cystoides intestinalis, has not yet been reported. Recently, a case of CCP mimicking pneumatosis cystoides intestinalis in the ascending colon was treated. Here this case is reported and the literature reviewed. (Intest Res 2010;8:187-190)