Diffuse Cavernous Hemangioma of the Colon and Rectum

A 40-year-old man was referred to our clinic for evaluation of long-standing, intermittent hematochezia, which was described as painless bright red blood per rectum that occurred with bowel movements. Past medical history was significant for a blood transfusion at the age of 13 for rectal bleeding. Physical examination was significant for spider angiomas on the chest. Laboratory testing revealed a hemoglobin level of 12.9 g/dL and MCV of 78 fL. A colonoscopy was performed, and subsequently a magnetic resonance image of the abdomen and pelvis was obtained.Colonoscopy (FigureA) revealed multiple confluent red-purple areas, suggestive of dilated vascular structures, extending from the rectum to 45 cm proximally. T2-weighted, coronal magnetic resonance images of the pelvis (FigureB) revealed circumferential thickening and increased signal in the distal sigmoid colon (FigureB, thick arrows) and entire rectum (FigureB, thin arrows), which possessed dilated vessels and phleboliths. Similar findings were present throughout the subcutaneous tissues of the penis. These findings were consistent with colonic hemangiomatosis. The patient subsequently underwent left hemicolectomy with colo-anal anastomosis. Surgical pathology confirmed the diagnosis of diffuse cavernous hemangioma involving the distal colon and rectum with marked, focal submucosal hemorrhage and multiple organizing thrombi involving the submucosa, serosa, and mesenteric fat (FigureC). The patient continues to do well 3 years after the surgery without recurrent hematochezia.Colonic hemangiomas are rare, nonmalignant lesions that arise from the submucosal vascular plexuses, as a result of embryonic sequestration of mesodermal tissue. Hemangiomas, histologically distinct from telangiectasias and angiodysplasias, may be classified as capillary, cavernous, or mixed.1Lyon D.T. Mantia A.G. Large-bowel hemangiomas.Dis Colon Rectum. 1984; 27: 404-414Crossref PubMed Scopus (77) Google Scholar, 2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar Cavernous hemangiomas, which comprise 70% of colonic hemangiomas, may occur as part of multisystemic or diffuse gastrointestinal angiomatosis, with rectosigmoid as the most common site of occurrence.2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar, 3Oner Z. Altaca G. Diffuse cavernous rectal hemangioma—clinical appearance, diagnostic modalities and sphincter saving approach to therapy: report of 2 and a collective review of 79 cases.Acta Chir Belg. 1993; 93: 173-176PubMed Google ScholarColonic hemangiomas can present as anemia, hematochezia, and even massive hemorrhage. Characteristic endoscopic findings are of soft, dilated, easily collapsible submucosal masses, ranging in color from deep wine to plum. A biopsy of these lesions should be performed with caution because massive bleeding can occur. Complete surgical excision of these lesions with a sphincter-saving procedure is the treatment of choice in symptomatic cases.2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar, 3Oner Z. Altaca G. Diffuse cavernous rectal hemangioma—clinical appearance, diagnostic modalities and sphincter saving approach to therapy: report of 2 and a collective review of 79 cases.Acta Chir Belg. 1993; 93: 173-176PubMed Google Scholar A 40-year-old man was referred to our clinic for evaluation of long-standing, intermittent hematochezia, which was described as painless bright red blood per rectum that occurred with bowel movements. Past medical history was significant for a blood transfusion at the age of 13 for rectal bleeding. Physical examination was significant for spider angiomas on the chest. Laboratory testing revealed a hemoglobin level of 12.9 g/dL and MCV of 78 fL. A colonoscopy was performed, and subsequently a magnetic resonance image of the abdomen and pelvis was obtained. Colonoscopy (FigureA) revealed multiple confluent red-purple areas, suggestive of dilated vascular structures, extending from the rectum to 45 cm proximally. T2-weighted, coronal magnetic resonance images of the pelvis (FigureB) revealed circumferential thickening and increased signal in the distal sigmoid colon (FigureB, thick arrows) and entire rectum (FigureB, thin arrows), which possessed dilated vessels and phleboliths. Similar findings were present throughout the subcutaneous tissues of the penis. These findings were consistent with colonic hemangiomatosis. The patient subsequently underwent left hemicolectomy with colo-anal anastomosis. Surgical pathology confirmed the diagnosis of diffuse cavernous hemangioma involving the distal colon and rectum with marked, focal submucosal hemorrhage and multiple organizing thrombi involving the submucosa, serosa, and mesenteric fat (FigureC). The patient continues to do well 3 years after the surgery without recurrent hematochezia. Colonic hemangiomas are rare, nonmalignant lesions that arise from the submucosal vascular plexuses, as a result of embryonic sequestration of mesodermal tissue. Hemangiomas, histologically distinct from telangiectasias and angiodysplasias, may be classified as capillary, cavernous, or mixed.1Lyon D.T. Mantia A.G. Large-bowel hemangiomas.Dis Colon Rectum. 1984; 27: 404-414Crossref PubMed Scopus (77) Google Scholar, 2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar Cavernous hemangiomas, which comprise 70% of colonic hemangiomas, may occur as part of multisystemic or diffuse gastrointestinal angiomatosis, with rectosigmoid as the most common site of occurrence.2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar, 3Oner Z. Altaca G. Diffuse cavernous rectal hemangioma—clinical appearance, diagnostic modalities and sphincter saving approach to therapy: report of 2 and a collective review of 79 cases.Acta Chir Belg. 1993; 93: 173-176PubMed Google Scholar Colonic hemangiomas can present as anemia, hematochezia, and even massive hemorrhage. Characteristic endoscopic findings are of soft, dilated, easily collapsible submucosal masses, ranging in color from deep wine to plum. A biopsy of these lesions should be performed with caution because massive bleeding can occur. Complete surgical excision of these lesions with a sphincter-saving procedure is the treatment of choice in symptomatic cases.2Hsu R.M. Horton K.M. Fishman E.K. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography.AJR Am J Roentgenol. 2002; 179: 1042-1044Crossref PubMed Scopus (29) Google Scholar, 3Oner Z. Altaca G. Diffuse cavernous rectal hemangioma—clinical appearance, diagnostic modalities and sphincter saving approach to therapy: report of 2 and a collective review of 79 cases.Acta Chir Belg. 1993; 93: 173-176PubMed Google Scholar The authors give special thanks to Evan S. Siegelman, MD, and Haohai Liang, MD, for providing radiologic and pathologic images, respectively.