TY - JOUR
T1 - Bleeding from cavernous angiomatosis of the rectum in Klippel-Trénaunay syndrome
T2 - Report of three cases and literature review
AU - Wilson, Candace L.
AU - Song, Louis Michel Wong Kee
AU - Chua, Heidi
AU - Ferrara, Marcello
AU - Devine, Richard M.
AU - Dozois, Roger R.
AU - Nehra, Vandana
PY - 2001
Y1 - 2001
N2 - Klippel-Trénaunay syndrome (KTS) is a congenital vascular anomaly characterized by limb hypertrophy, cutaneous hemangiomas, and varicosities. GI hemorrhage is a potentially serious complication secondary to diffuse hemangiomatous involvement of the gut. We report on three patients with KTS who presented with transfusion-dependent anemia and life-threatening bleeding due to extensive cavernous hemangiomas involving the rectum. Two patients were treated by proctocolectomy and coloanal anastomosis, which preserved anal function while controlling bleeding. The third patient required an abdominoperineal resection because of extensive rectal, perianal, and perineal angiomatosis. The literature on the evaluation and management of GI hemorrhage in KTS, particularly of colorectal origin, is reviewed.
AB - Klippel-Trénaunay syndrome (KTS) is a congenital vascular anomaly characterized by limb hypertrophy, cutaneous hemangiomas, and varicosities. GI hemorrhage is a potentially serious complication secondary to diffuse hemangiomatous involvement of the gut. We report on three patients with KTS who presented with transfusion-dependent anemia and life-threatening bleeding due to extensive cavernous hemangiomas involving the rectum. Two patients were treated by proctocolectomy and coloanal anastomosis, which preserved anal function while controlling bleeding. The third patient required an abdominoperineal resection because of extensive rectal, perianal, and perineal angiomatosis. The literature on the evaluation and management of GI hemorrhage in KTS, particularly of colorectal origin, is reviewed.
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U2 - 10.1016/S0002-9270(01)02673-9
DO - 10.1016/S0002-9270(01)02673-9
M3 - Article
C2 - 11569713
AN - SCOPUS:0034834582
SN - 0002-9270
VL - 96
SP - 2783
EP - 2788
JO - American Journal of Gastroenterology
JF - American Journal of Gastroenterology
IS - 9 SUPPL.
ER -