An Unusual Case of Lower Gastrointestinal Hemorrhage

Question: A 53-year-old woman, who was in good health, presented with hematochezia for more than 20 days. Examination was unremarkable, with stable vital signs. Laboratory tests revealed a hemoglobin of 78 g/L (normal 120–160 g/L), and the rest of the laboratory tests were normal. Colonoscopy revealed multiple masses with a cobblestone appearance, obvious mucosal congestion, and edema in the junction of sigmoid and rectum (Figure A). Colonic biopsy precipitated active bleeding that required various measures to stop bleeding. Ice-cold saline solution and 8% norepinephrine (8 mg norepinephrine in 100 mL saline solution) was used to rinse the bleeding site repeatedly and to constrict the vessels, but failed to stop the bleeding. Titanium clip was then used to clamp the vessels, but blood still exudated. Lauromacrogol was injected to stop the bleeding eventually (Figure A). She recovered following fluid resuscitation, acid suppression and somatostatin therapy. Contrast-enhanced computed tomography (CT) of the abdomen and pelvis is shown in Figures B–E. Three days later, she developed massive hematochezia with a hemoglobin level of 45 g/L. Owing to effective fluid resuscitation and blood transfusion, her blood pressure was relatively stable. What is the most likely cause for lower gastrointestinal hemorrhage in this patient? See the Gastroenterology website (www.gastrojournal.org) for more information on submitting to Gastro Curbside Consult. In this case, cobblestone appearance and obvious mucosal congestion (Figure A) made us suspect inflammatory bowel disease (IBD). However, bleeding following biopsy indicated other possibilities. We next performed CT to get some clue. Also, a CT scan could help to assess the extent of the lesions. The axial CT plain scan image (Figure B) suggested irregular thickening and local luminal narrowing of the colonic walls in the left upper abdomen. These colonic lesions showed bead-like and tortuous tubular enhancement in the arterial phase (Figure C), and their density continued to enhance in the portal phase (Figure D). On the coronal portal phase image (Figure E), multiple nodular soft tissue-density lesions, which showed similar enhancement degree as that of adjacent large vessels, were observed around the hilar of liver, abdominal aorta, inferior vena cava, pancreas, and pelvic cavity; in addition, more extensive intestinal lesions (ie, transverse colon, descending colon, and sigmoid) were observed. These signs did not support IBD, but indicated extensive cirsoid aneurysm of the mesentery, retroperitoneum, and colon. Finally, we checked the histology of the biopsy samples (Figure F), which showed vascular proliferation in the submucosa of the intestinal wall (black arrows). Some vascular lumina were inconspicuous (green arrowhead), whereas others contained red blood cells (blue arrowhead), suggesting cirsoid aneurysm. Cirsoid aneurysm is an arterio-venous malformation with unknown etiology. Cirsoid aneurysms of the stomach and jejunum have been reported to cause gastrointestinal hemorrhage,1Eidus L.B. Rasuli P. Manion D. et al.Caliber-persistent artery of the stomach (Dieulafoy’s vascular malformation).Gastroenterology. 1990; 99: 1507-1510Abstract Full Text PDF PubMed Scopus (36) Google Scholar,2Vetto J.T. Richman P.S. Kariger K. et al.Cirsoid aneurysms of the jejunum. An unrecognized cause of massive gastrointestinal bleeding.Arch Surg. 1989; 124: 1460-1462Crossref PubMed Scopus (38) Google Scholar whereas cirsoid aneurysms in the colon are rarely described. CT angiography is a valuable diagnostic tool, and treatment options depend on the lesions.3ElKiran Y.M. Abdelgawwad M.S. Abdelmaksoud M.A. et al.Surgical management of cirsoid aneurysms of the scalp: ten years’ experience.World Neurosurg. 2021; 150: e756-e764Crossref PubMed Scopus (4) Google Scholar In the present case, the lesions were extensive and surgery was contraindicated. Interventional embolization proved to be a good option. In addition, biopsy needs to be performed cautiously, especially for those with obscure gastrointestinal bleeding. The patient underwent emergency angiography, which revealed extravasation of contrast in the junction of sigmoid and rectum, and embolization of the superior rectal artery was performed (Figure G). She had no further active bleeding after the embolization. During follow-up, the patient was relatively stable. CT angiography was performed 5 years later. The 3-dimensional image revealed the extensive cirsoid aneurysm (Figure H), which was consistent with the previous CT findings. She was suggested to have a regular visit and seek a doctor immediately if melena or abdominal discomfort occurs.