Abstract
BackgroundColitis cystica profunda is a rare nonneoplastic disease defined by the presence of intramural cysts that contain mucus, usually situated in the rectosigmoid area, which can mimic various malignant lesions and polyps. Its etiology still remains not fully elucidated, and several mechanisms such as congenital, post-traumatic, and infectious have been implicated in the development of this rare entity.Case presentationHerein, we describe a unique case of colitis cystica profunda in the setting of Peutz-Jeghers-type polyp of the sigmoid colon, associated with high-grade dysplasia of the overlying epithelium in a 48-year-old female patient, who presented to the emergency room with signs of intestinal obstruction. To the best of our insight, this is the first manifestation ever reported in the literature regarding the coexistence of solitary Peutz-Jeghers-type polyp, colitis cystica profunda, and high-grade dysplasia of the epithelium of the colon.ConclusionsThe purpose of this case report is to highlight colitis cystica profunda and its clinical significance. An uncommon nonneoplastic entity, many times masquerading as malignant lesion of the rectosigmoid area of the colon. Clinicians and pathologists should be aware of this benign condition that is found incidentally postoperatively in patients undergoing colectomies, leading to unnecessary increase of morbidity and mortality in these patients, who otherwise could have been cured with conservative treatment only.
Highlights
Colitis cystica profunda is a rare nonneoplastic disease defined by the presence of intramural cysts that contain mucus, usually situated in the rectosigmoid area, which can mimic various malignant lesions and polyps
Many times masquerading as malignant lesion of the rectosigmoid area of the colon
cystica profunda (CCP) is a nonmalignant lesion defined by submucosal mucin-occupying cysts of variable size, which are covered by an epithelium with no cellular atypia and extend below the muscularis mucosa, intruding into the muscularis propria of the colonic wall in many cases
Summary
Knowledge of CCP and its clinical presentation are indispensable to differentiate this benign entity from malignancy. There are cases in which this uncommon entity overlaps with dysplastic changes of the epithelium of the bowel, a fact that needs extra attention from the medical practitioners and from the pathologists who examine the histology of the disease. The association with other rare entities like solitary Peutz-Jeghers-type polyp, as in our case, is of paramount importance in order to avoid overly aggressive treatment decisions. Based on the current medical literature, our reported case is unique, highlighting the potential association of CCP with dysplasia along with the coexistence of solitary hamartomatous polyp; an association that has never been reported in the past
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