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REVIEW Ectopic Variceal Bleeding 388 REVIEW Ectopic variceal bleeding due to portosystemic shunt via dilated mesenteric veins and a varicous left ovarian vein : case report and literature review of ectopic varices M. Struyve1,2, G. Robaeys1,2 (1) Department of Gastroenterology and Hepatology, University Hospitals Gasthuisberg, Leuven, Belgium ; (2) Department of Gastroenterology and Hepatology, Ziekenhuis Oost-Limburg (ZOL), Genk, Belgium. Abstract 400]), a decreased prothrombin time (68%), a normal kidney function (serum creatinine level 0.93 mg/dl [0.5- Ectopic varices are dilated portosystemic venous collaterals 0.9]), no signs of inflammation, normal liver enzymes located outside of the gastro-esophageal region. Whereas they are common endoscopic findings in patients with portal hypertension, and a hyperammonia (356 microgram% (11-51)). ectopic variceal bleeding is rather rare and accounts for only 1 to An urgent esofagogastroduodenoscopy (EGD) was 5 % of all variceal bleedings. The rectum and the duodenum are performed bedside at the ICU after stabilization of the the most common sites for ectopic varices, but they can be present along the whole intestinal tract and neighborhood. At present, patient. Neither an active bleeding nor potential bleeding there is no consensus well established on diagnostic workup for source could be found. A left colonoscopy immediately ectopic variceal bleeding and their therapeutic strategies. Further followed showed a massive active lower intestinal investigation of large series or randomized-controlled trials is needed because nowadays most of the data available are based on bleeding, but again the site of bleeding could not be case reports. We report here an unusual case of an ectopic variceal discovered. bleeding, presented as an acute small intestine bleeding, due to a A contrast-enhanced computed tomography (CT) scan portosystemic shunt via dilated mesenteric veins and a varicous left ovarian vein in a patient with alcoholic cirrhosis. The involvement of the abdomen was performed shortly after (Fig. 1-3). of an ovarian vein in ectopic variceal bleeding is rarely described. This examination demonstrated advanced cirrhosis with (Acta gastroenterol. belg, 2017, 80, 388-395). signs of portal hypertension (PHT), i.e. splenomegaly, Key words : Ectopic varices, ovarian vein, portal hypertension, edema in the mesenteric fat, some ascites and varices. intestinal bleeding, cirrhosis. Furthermore a spontaneous portosystemic shunt was demonstrated, occurring by connections between Abbreviations : ICU, intensive care unit EGD, esofago- mesenteric veins and a varicous left ovarian vein (Fig. 1). gastroduodenoscopy – CT, computed tomography – PHT, portal hypertension – EV, ectopic varices – TIPS, transjugular intrahepatic portosystemic shunt – MRI, magnetic resonance imaging – B-RTO, balloon occluded retrograde transvenous obliteration – MELD, model of end stage liver disease CASE REPORT A 53-year-old woman was admitted at a psychiatric facility because of alcohol abuse. The patient was found at night lying unconsciously on the floor in a large pool of blood. She was urgently transferred to the hospital under medical assistance where she was stabilized and intubated and afterwards transported to the Intensive Care Unit (ICU). Her medical history consisted of an important addiction of alcohol with diagnosis of alcoholic cirrhosis in 2005 and schizophrenia. A physical examination at Fig. 1. — CT-angiographic reconstructions demonstrating a admission revealed a tension of 112/63 mmHg, a pulse broad portosystemic shunt via dilated mesenterial and ovarian of 83 beats per minute and an oxygen saturation ratio veins, communicating with eachother. of 99% with a FiO2 of 21%. Glasgow Coma Scale was 7/15. The abdomen was slightly tender. Rectal digital examination showed massive melena mixed with dark Correspondence to : Mathieu Struyve, Department of Gastroenterology and blood cloths. Hepatology, University Hospitals Leuven, Herestraat 49, 3000 Leuven, Belgium E-mail : [email protected] Laboratory analyses demonstrated a deep anaemia (a serum hemoglobin of 5 g/dl [11.7-16.1]), a mild Submission date : 03/01/2017 thrombopenia (blood platelets 141 x 1000/mm³ [150- Acceptance date : 02/02/2017 Acta Gastro-Enterologica Belgica, Vol. LXXX, July-September 2017 Struyve.indd 388 3/10/17 14:20 Ovarian ectopic variceal bleeding 389 arise from capillary complexes that form a submucosal venous plexus in the wall of the gastrointestinal system and drain into short veins penetrating the muscular intestinal layer subsequently into major veins, forming an extensive communicating venous network. Intestinal varices are defined as dilated blood vessels in this network, occurring in case of increased portal pressure. Deep intrinsic veins in the mucosa of the gastrointestinal tract will enlarge and become tortuous variceal channels. Also an increase in number and size of the venous vessels in the lamina propria will occur (4). They may shunt blood from the portal circulation to the systemic venous circulation, if communication between varicous portal veins and systemic veins occur. The most common sites are the esophagus and the gastric fundus. Ectopic varices are considered as dilated portosystemic Fig. 2. — CT image shows a dilated and varicous left ovarian collateral veins along the digestive tract other than the vein, communicating with dilatated mesenteric veins (not esophageal and gastric region. The duodenum, small shown). intestine, colon, rectum, peritoneum, abdominal wall, These ectopic varices (EV) were in close contact with a the submucosa of a dilated segment of the ileal part of the small intestine (Fig. 3). In addition, CT demonstrated profound active intestinal bleeding caused by part of these ileal varices (Fig. 3). During the short stay at the ICU, there was a hemodynamic stabilization of the patient with a spontaneous cessation of the intestinal bleeding, observed by meaning of stabilization of the serum hemoglobin value and spontaneous resolution of melena. In contrast, there was a progressively and rapid neurological detoriation due to post-hypoxic cerebral edema with refractory epileptic insults. Therefore we were not able to perform any endoscopic interventional treatment, interventional radiological procedures (transjugular intrahepatic portosystemic shunt (TIPS) e.g.) nor surgical treatment to manage the ectopic varices. The patient unfortunately died due to neurological complications. b LITERATURE REVIEW We report here an unusual case of an ectopic variceal bleeding, presented as an acute small intestine bleeding, due to a portosystemic shunt via dilated mesenteric veins and a varicous left ovarian vein as a sign of PHT in the setting of alcoholic cirrhosis. A literature search on PUBMED from 1958 to 2016 using keywords ectopic varices showed 186 hits but the involvement of the ovarian veins was only in three case reports described (1-3). Pathogenesis and etiology The portal venous system is a complex and extensive Fig. 3. — Computed tomography (CT) image demonstrating tract that drains blood from parts of the gastrointestinal contrastleakage in a dilated segment of the small intestine (a,b). system, the pancreas and the spleen to the liver. The (b) CT image in the portal-venous phase demonstrating further gastrointestinal drainage occurs from the lower part of the intraluminal dilution of the intraluminal contrastleakage as an esophagus to the upper part of the anal canal. These veins indicative sign of an acute and active intestinal bleeding. Acta Gastro-Enterologica Belgica, Vol. LXXX, July-September 2017 Struyve.indd 389 3/10/17 14:20 390 M. Struyve et al. retroperitoneal space and even the ovary (like in our the next step to perform if endoscopy fails to identify the case) are potential sites of ectopic varices. bleeding source (5,8,10-12). Luminal ectopic varices (within the gastrointestinal Spiral CT scan is optimally performed in multiple tract lumen) are distinguished from non-luminal ectopic phases. Non-contrast scan is followed by scanning the varices (elsewhere in the abdominal cavity or the pelvis) entire abdomen in the arterial and portal venous phase and the latter can be more difficult to detect (5). The most of contrast perfusion. Furthermore, in case of suspected frequent sites of ectopic varices are the rectum (44.5 %) intra-abdominal hemorrhage it is important that the and the duodenum (32.9 %). The ovaries, like in this patient is not prepared with a positive (gastrografin, case report, are an infrequent site for ectopic varices (5- barium) peroral contrast, otherwise intraluminal contrast 6). These findings were confirmed by Norton et al. in a extravasation can be easily missed. Elective capsule review of 169 cases of bleeding due to ectopic varices endoscopy is succesfull in detecting the presence of small (7). Whereas ectopic varices are relatively common bowel varices, however without any therapeutic potential findings during endoscopy in PHT, ectopic variceal in acute bleeding (5,8,13,14). Color Doppler ultrasound bleeding however are rare and counts for only 1 to 5 % is another technique that has proven to be helpful in of all variceal bleeding episodes (1,7,8). the diagnosis. However, topography of the extent of In normal and healthy conditions, portosystemic the varices is best demonstrated by CT or Magnetic collaterals are anatomically present but the portal Resonance Imaging (MRI) (5,8,15-18). Double balloon bloodstream flows by preference in a hepatopetal enteroscopy is useful as well in
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