Incidental finding of diffuse cavernous rectal haemangiomatosis during bowel cancer screening

Abstract

BackgroundThis case seeks to highlight to endoscopists a rare benign disorder that may be encountered during endoscopy. Clinicians may be tempted to biopsy, which could lead to a catastrophic gastrointestinal haemorrhage.Case presentationA 66-year-old asymptomatic Caucasian male was referred for colonoscopy with a positive faecal occult blood test as part of the UK national bowel cancer screening programme. Relevant past medical history included atrial fibrillation for which he took Dabigatran. He had a normal haemoglobin, mean cell volume, platelet and clotting function.During colonoscopy, an unusual vascular pattern encompassing the entire rectum extending to the rectosigmoid junction was noted at intubation. The lesion demonstrated confluent circumferential purple discolouration indicating venous blood supply, with heaping up of the mucosa involving the entire rectum and rectosigmoid junction. There was no corresponding history of venothromboembolic disease or liver disease. The patient proceeded to have computed tomography (CT) which revealed a considerably thickened rectosigmoid wall with multiple small rounded punctate calcifications within it, and no other visceral involvement. Subsequent magnetic resonance (MR) scan of the pelvis demonstrated extensive diffuse thickening of the rectum and lower sigmoid with intermediate to high T2 signal, and an internal architecture of multiple ‘grapelike’ lobulations.ConclusionThe findings were consistent with diffuse cavernous haemangiomatosis of the rectum (DCHR), an extremely rare benign submucosal vascular intestinal tumour originating from the dentate line. Misdiagnosis of DCHR is common and the macroscopic appearance of DCHR can mimic varices, haemorrhoids, polyps or proctitis. MR imaging is the gold standard for diagnosis. Common presentation is with haematochezia due to mucosal wall erosion. The treatment of choice for symptomatic DCHR is pull-through transection and colo-anal anastomosis. This case seeks to highlight a rare disorder that can be encountered incidentally during lower GI endoscopy. Injudicious biopsy is potentially catastrophic. In a patient who endoscopically has evidence of a DCHR, we advocate MR pelvis assessment to clarify the nature of the lesion to guide future management if required. The patient discussed remains well, asymptomatic, with no evidence of iron deficiency anaemia.

Revisiting imaging features of rectosigmoid vascular malformation with emphasis on multiparametric MRI: a case report

“…Abeysekera et al demonstrated CT and MRI findings of a patient with an incidentally detected rectal vascular malformation in an older man. In their report, the CT scan depicted wall calcifications, and the MRI scan showed diffuse T2W hyperintense wall thickening accompanied The imaging findings provided by Abeysekera et al bear a close resemblance to the findings in our case, except for the lack of early nodular contrast enhancement [9]. Furthermore, their patient was asymptomatic and did not undergo an operation, while the patient presented in this report had typical symptoms of diffuse rectosigmoid vascular malformation and clinical history and underwent an operation.…”

“…Most of these papers were published before the 2010s; thus, the imaging findings were inevitably restricted to that era's technology. Nonetheless, some of these reports were published last decade [4,[9][10][11]. In their large sample of children with vascular malformation of the colorectum, Peng et al showed the yields of ultrasonography in the diagnosis [4].…”

Laparoscopic total mesorectal excision for rectal venous malformation: A case report with a brief literature review

“…Biopsy of these lesions is not recommended because it can lead to severe bleeding. 18 With regard to imaging, both CT angiography and MRI were performed in the present case. CT angiography can characterize the disease and delineate the extent and involvement of adjacent structures.…”

“…In our case, colonoscopy and imaging were diagnostic. Biopsy of these lesions is not recommended because it can lead to severe bleeding 18 …”