Endosonographic Findings in Colitis Cystica Profunda.

Case Report

A 27-year-old male was referred to our institution for further evaluation of persistent rectal bleeding. A prior colonoscopy showed a sigmoid soft tissue lesion, and pathology revealed chronic active colitis and granulation tissue with ulcers and focal adenomatous changes. We performed a flexible sigmoidoscopy that showed a 4.5-cm multilobulated polypoid lesion approximately 45 cm from the anal verge (Figure 1). A 20 MHz Olympus endoscopic ultrasound (EUS) miniprobe showed hypoechoic lesion with cystic/spongy features involving the mucosa and submucosa (Figure 2). These features were thought to suggest colitis cystica profunda (CCP). Histologic examination of snare biopsies identified dilated glands with mucinous content, surrounded by variable degrees of fibrosis on a background of interspersed chronic inflammatory cells, with few colonic mucosal crypts and mild inflammatory cell infiltrate (Figure 3). The patient was instructed to follow up with gastroenterology if bleeding recurred.

Figure 1

Endoscopic image of CCP showing polypoid lesion with lobulated appearance and some hemorrhagic areas with normal surrounding mucosa.

Figure 2

EUS showing hypoechoic lesion with cystic/spongy features, with involvement of the mucosa and submucosal. The muscularis propria appears intact.

Figure 3

Hematoxylin and eosin stain section showing transmural dilated glands with mucinous content, surrounded by variable degrees of fibrosis on a background of interspersed chronic inflammatory cells and few colonic mucosal crypts, with mild inflammatory cells infiltrate.

CCP is a rare, benign disease of the colon and rectum often mimicking malignancy. It was first described in 1766 by Stark, who reported 2 cases associated with dysentery. Histologically, it is characterized by dilated mucous glands mostly limited to the submucosa, but there are reported cases of penetration to the muscularis mucosa. The etiology of CCP remains controversial; however, many consider solitary rectal ulcer syndrome (SRUS) and CCP to be different manifestations of the same pathology due to overlapping features. Surface mucosal biopsies may rule out neoplasia, but only deep biopsies show characteristic histological features. There are few case reports of the endosonographic features of CCP, which include multiple hypoechoic or anechoic lesions affecting mucosa or submucosa, with areas of echorefringent fibrosis between lesions in the absence of lymph node enlargement. EUS was instrumental in making our diagnosis and providing proper counseling to the patient.

Disclosures

Author contributions: All authors contributed equally to this article. M. Sultan is the article guarantor.

Financial disclosure: The authors have no financial disclosure.

Informed consent was obtained for this case report.