Diffuse cavernous hemangioma of the rectum: case report and literature review.

Abstract

Dear Editor: Gastrointestinal hemangiomas and vascular malformations are uncommon benign vascular tumors that can occur anywhere in the gastrointestinal tract. They can be distributed throughout the intestinal digestive system or present as a single cavernous hemangioma/malformation, which is often located in the rectosigmoid region [1]. We report a case of a 44-year-old woman who was previously asymptomatic and had a normal physical examination, and laboratory parameters underwent a colonoscopy after a single episode of rectal bleeding. The endoscopic evaluation revealed, in addition to a 30-mm sessile polyp resected at that time, the presence of an extensive, purplish, embossed lesion, with focal aberrant vessels occupying the rectum. Given the suspicion of a vascular origin of the lesion, no biopsy samples were taken. Narrow band imaging (NBI) helped us to establish the boundaries of the polypoid lesion, and with careful positioning of the polypectomy snare, we avoided the resection of other pseudopolypoid hemangiomatous lesions. A contrastenhanced MRI showed a heterogeneous mass of 7.5×3.5 cm in size in the posterior wall of the rectum, with high signal intensity on T2-weighed images and multiple areas of low signal intensity on T1 and T2, possibly related to phleboliths. The mass had the lower extremity at the level of the rectal insertion of the elevator muscle of the anus, avoiding the anal canal and adjacent structures. The study was completed with endoscopic ultrasonography that confirmed the presence of a heterogeneous hypoechoic lesion, containing anechoic and hyperechoic areas, limited by muscularis propria. The lesion had pulsatile flow by Doppler study and was supplied by a small extraluminal vessel. An upper endoscopy and an angiography were performed to exclude the presence of other gastrointestinal vascular lesions. Given the possibility of recurrent bleeding with possible severe hemorrhage (even occult) and the size of the lesion, a surgical procedure was discussed with the patient, who decided to postpone the procedure. Phillips first described cavernous hemangioma of the large intestine in 1839 [2]. Since then, over 200 cases of colonic cavernous hemangiomas and approximately 100 cases of diffuse cavernous hemangioma of the rectum (DCHR) have been reported. An estimated 80 % of patients with DCHR exhibit symptoms, with intraluminal bleeding in the majority of cases. Up to 90 % have recurrent painless bleeding; half will have chronic iron-deficient anemia [3]. Other symptoms stem from a possible compression or invasion of adjacent structures, such as lumbar or perianal pain, metrorrhagia, and hematuria [4]. Misdiagnosis is a major theme in cases of hemangiomas and malformations. A large number of these lesions are nearly missed or falsely diagnosed because symptoms are attributed to hemorrhoids, polyps, or ulcerative colitis, resulting in an average delay of diagnosis of approximately 19 years. Furthermore, approximately 80 % of patients undergo one prior inappropriate surgical procedure, typically hemorrhoidectomy [3, 4]. Our case is an example of an atypical presentation because the patient remained asymptomatic and without anemia until adulthood. Colonoscopy is undoubtedly the diagnostic technique of choice, allowing for the assessment of localization, morphology, and total extension of the lesion [4]; DCHR typically appears as soft and compressible bluish or deep red submucosal lesions, with dilated and engorged veins in the rectal wall. * Patricia Andrade anapatriciarandrade@gmail.com