Abstract

        Splenic Artery Aneurysms (SAAs) are the most common type of abdominal visceral artery aneurysm. Most of the splenic artery aneurysm is asymptomatic, being found incidentally at the time of investigations or surgery. Complications of SAAs include free intraperitoneal rupture and erosion into abdominal viscera or vessels, such as pancreatic duct, esophagus, stomach, or large bowel, which can results in gastrointestinal bleeding or arteriovenous fistula and associated with high morbidity and mortality. Treatment of such aneurysms depends on the patient general condition, presentation and location of the aneurysm in relation to the spleen, and the known morbidities for each procedure. Treatment options include open surgery, laparoscopic surgery, and endovascular stent graft and transcatheter embolization. The present case is the first, to our knowledge, of true SAA presenting with massive lower gastrointestinal bleeding from erosion into colon treated successfully with transcatheter arterial embolization with no recurrent of gastrointestinal bleeding in ten months follow-up.

Case Report

A 78-year-old male present to the Emergency Department with massive rectal bleeding left lateral flank pain and a left lower quadrant pain for five days after he was found on the floor surrounded in a pool of blood at his nursing home. He describes frank red blood per rectum but no change in his bowel habit. He had no nausea, vomiting or weight change. His past medical history includes chronic obstructive pulmonary disease, hypertension, duodenal ulcer, hypercalcaemia, cholelithiasis, small abdominal aortic aneurysm measuring 3cm, small splenic artery aneurysm measuring 1.9cm, and alcohol abuse for more than ten years and pancreatitis. A colonoscopy done two years earlier was normal. On examination he was afebrile with an oxygen saturation of 97% on room air and hemodynamically stable initially but he became tachycardic and hyoptensive (heart rate of 104 per minute and blood pressure of 80/60 mmgh) while he is being evaluated. He was pale and had left lower quadrant tenderness on deep palpation. Digital rectal examination revealed frank bright red blood. The first haemoglobin result was 108g/L and coagulation profile was normal. Rapid fluid resuscitation started in the form of 3 litres of normal saline as well as three units of blood, and his associated blood work showed 4 grams haemoglobin drop. Once stabilize he was transferred to the angiography suit. Through right common femoral puncture. A catheter and guide wire advanced with some difficulty through markedly diseased iliac systems with a combination of stenoses and aneurysms in the abdominal aorta, which is also aneurysmal. Three injections were made into the aorta, which confirmed diffuse atheromatous change with aneurysmal dilatation. The superior mesenteric artery (SMA) vessel origin looks normal, limited filling of the celiac origin and no aortoduodenal fistula (this was the main clinical concern)
Figure 1.

 

 Selective injection into the SMA was made which showed no bleeding point Figure 2.

 

 A limited celiac injection  was made because of catheter flipping back into the aorta. There was, in retrospect an enhancing focus in the region of the splenic artery which subsequently turned out to be an aneurysm Figure 3.

 

Since we had not nailed the diagnosis the patient was taken to CT for a CT angiography. Contrast was injected at 5 cc a second with 3 second delay. There was an enhancing aneurysm of the splenic artery which communicates with the lumen of the colon Figure 4.
 

This was the obvious source of the bleeding. A 4 cm abdominal aortic aneurysm is also seen below the origins of the renal arteries Figure 5

 

. In view of the findings, the patient was returned to the angiogram suite for embolization. Catheter and guide wire combination were advanced into the celiac axis with great difficulty in view of the tortuosity and the status of the vessels. A micro catheter was advanced well into the splenic artery but we were not able to get into the distal spleen artery to occlude "back-door" of the aneurysm. We therefore deployed three coils in the proximal splenic artery. This caused complete cessation of flow. We stopped at that point and removed the catheter with no immediate complication. A follow-up CT showed a 5 cm splenic artery aneurysm close to the splenic hilum which totally thrombosed and a few air bubbles are noted within this secondary to fistula between the splenic aneurysm and the colon Figure 6.

 

There was also decreased density in the major part of the spleen suggesting splenic infarct. Postprocedural the patient was transferred to the ICU for further monitoring and observation. While in the ICU he developed chest pain but no myocardial infarction. He tolerated an advancing diet and remained on intravenous antibiotics for one week. He was discharge back to the nursing home one week later on oral antibiotic for six weeks. He remains symptoms free on ten months follow-up.



Materials and Methods


         Reports of Splenic artery aneurysm, pseudoaneurysms and gastrointestinal haemorrhage or bleeding identified through a Medline database search from 1966 to August 2005 using Ovid software (Ovid Technologies, Inc, New York, NY) and the search strategy [Splenic artery and aneurysm pseudoaneurysms (Exploded MeSH headings)] and [gastrointestinal haemorrhage or bleeding]. Reference lists of all relevant articles and reference lists of review articles were also examined.
 

Review of the Literature


        Thirty-six cases of splenic artery aneurysms ruptured into the stomach been reported from 1963 until August 2005 (Table 1),
 

sixteen cases of splenic artery aneurysms ruptured into the colon (Table 2)

 

and three cases of splenic artery aneurysms ruptured into both stomach and colon (Table 3)
 
 

 making a total of 55 cases. All presented with gastrointestinal bleeding. Of these, 20 had true splenic artery aneurysms (38%) (Sixteen in the first group splenic artery aneurysm rupture into the stomach (1-10) and four in the second group in which the splenic artery aneurysm rupture into the colon (11-14) and 31 had history of pancreatitis or alcohol abuse (56%). Thirty patients were male (55%), 13 female (23%), and in twelve the sex was undetermined (22%). Eleven out of thirteen female had true splenic artery aneurysms (85%) (1, 2, 4, 5, 7-10, 12, 13) were as eight out of thirty male had true splenic artery aneurysms (26%) (3, 11, 14, 15). The age ranged from 30 to 83 years for both sex, with a median of 49 years. The median age for the female patients were 62 years and for the male patients 49 years. Median size of the true splenic artery aneurysm was 3 cm (range, 2-7cm) and for the pseudoaneurysm 6.5 cm (range, 2-15cm).


Management



       Of the 55 cases reported, in five cases both the management and the outcome are not described (16-20) and in another two cases, one the managing (21) and the second one the outcome (22) is not described but we include them in the tables. Four did not undergo surgery, either because they died before or because deemed at too high risk. All of them died of haemorrhage (4, 15, 23, 24). Twenty-two out of the 41(54%) who had surgery ended with splenectomy (1-3, 5, 7, 8, 11-14, 25-36). Seven (15%) were treated with transcatheter arterial embolization (11, 25, 37-41) two out of seven failed (29%) and had surgeries (11, 25) all for splenic artery pseudoaneurysms. Ten (24%) had ligation of the splenic artery aneurysms with out splenectomy (3, 6, 9, 21, 42-44). The mortality in patients presented with splenic artery aneurysm rupture into the stomach is 20%, 37.5% into the colon and 67% into both stomach and colon. Patient with pseudoaneurysm had higher mortality than the one with true splenic artery aneurysm (31% Vs 21%). The overall mortality is 29%. All the patients who had the transcatheter arterial embolization survived.



Discussion

 

        Since Beaussier (45) first description of splenic artery aneurysms (SAAs) in 1770, SAA are the most common type of abdominal visceral artery aneurysm accounting for 58% (29) and the third most common aneurysm in the abdomen second only to those of the infrarenal aortic and iliac arteries (5). The true incidence of splenic artery aneurysms is unknown and their prevalence varies widely. Ranging from 0.01% to 0.23% in autopsy studies, however the prevalence is much higher (10.4%) in autopsy limited to elderly(46), to 0.78% in a radiology study (29). Women are four times more likely than men to develop these aneurysms (29). In our review, the incident of splenic artery aneurysm higher in male than female (55% and 23% respectively) but this due to the higher percentage of pseudoaneurysms in the male patients comber to the female (73% Vs 15%).
          Four distinct factors may contribute to the development of splenic artery aneurysms, including two that account for their unusual female predilection. The first factor is medial fibrodysplasia, which usually occurs in women and is most often manifested by renal artery stenosis and secondary hypertension. 4% of patients with dysplastic renal artery disease have splenic artery aneurysms and all of these patients have been women. The second factor relates to the deleterious consequences of pregnancy, with its known increase in splenic blood flow and reproductive hormone-related changes in elastic vascular tissue. The third factor is evident in the nearly 10% of patients with portal hypertension and splenomegaly who develop splenic artery aneurysms. The forth factor focal arterial inflammatory processes due to septic emboli or pancreatitis. Nearly all splenic artery aneurysms associated with arterial fibrodysplasia, multiple pregnancies, or portal hypertension are saccular and occur at branching. At such sites, discontinuities exist in the internal elastic lamina of normal vessels, and any subsequent degenerative events involving elastic tissue, as might occur with arterial fibrodysplasia or pregnancy, are likely to produce aneurysmal changes. Splenic artery branch aneurysms are multiple in 20% of cases. Proximal aneurysms of the main splenic artery are usually solitary and are frequently associated with pancreatitis-related pseudocysts (29). In our review, 55% of patients had history of pancreatitis or alcohol abuse. Pancreatitis-related aneurysms are not usually associated with intraperitoneal bleeding; more often, they are a source of intestinal hemorrhage after rupture into the stomach or pancreatic ductal system (7).
        Generally splenic artery aneurysms are asymptomatic and usually discovered incidentally on abdominal imaging. Rarely, a tender, pulsatile mass in the upper abdomen may be palpated. The most common complication in the symptomatic patient is vague pain (29) other complications include rapture into the peritoneal cavity, erosion and rupture into splenic vein (producing portal hypertension), pancreatic duct (producing hemoductal pancreatitis) or the gastrointestinal tract (producing gastrointestinal haemorrhage). The patients typically present with acute abdominal pain and shock. The risk of splenic artery aneurysm rupture depends on a number of confounding and poorly defined factors. In general, rupture of bland aneurysms occurs in the range of less than 2% to 3% of cases (7,29). Rupture appears just as likely when the aneurysm is calcified, occurs in a normotensive patient, or occurs in the very elderly patient. Pregnancy is a major risk factor; nearly 95% of aneurysms recognized during pregnancy have ruptured (29). The maternal mortality rate approaches 75%, and the fetal mortality rate exceeds 95% in these cases (47). Pregnancy-related rupture occurs most often during the third trimester (69%) and is less common during the first two trimesters (12%), during labour (13%), or postpartum (6%) (48). In our review, we find two factors effects the mortality first the location of rupture of the splenic artery aneurysm into the gastrointestinal tract (20% when it rupture into the stomach, 37.5% into the colon and 67% into both stomach and colon) and second the type of splenic artery aneurysm (pseudoaneurysm Vs true splenic artery aneurysm 31% Vs 21%) patient general condition and the higher incident of pancreatitis may contribute to the higher mortality. The mortality is much lower (5%) in patient present with gastrointestinal hemorrhage secondary to hemorrhage into the pancreatic duct from a splenic artery aneurysm, Harper et al (49) reviewed 20 such cases surgery was curative in 18 cases, one treated with embolization and the only one fatality reported was in a patient who refused surgery. Indications for therapy for SAAs include presence of symptoms, presence in women of childbearing age, size more than 1.5cm especially in patient with portal hypertension, and documented enlargement of the aneurysm.
         Lindboe (50) in 1932 made the first preoperative diagnosis of SAA, followed by successful operative management and in 1978 Probst (51) reported the first case of transcatheter arterial embolization of splenic artery aneurysm. Matsumoto (39) reported the first case of a successful laparoscopic repair of splenic artery aneurysm in 1997. Current methods of management for SAA include transabdominal open or laparoscopic surgery, endovascular stent graft and transcatheter embolization of the aneurysm. Methods of transabdominal surgery include splenectomy with removal of the SAA, proximal and distal SAA ligation with or without aneurysmectomy. Of the 55 cases reported, in five cases both the management and the outcome are not described (16-20) and in another two cases, one the managing (21) and the second one the outcome (22) is not described but we include them in the tables. Four did not undergo surgery, either because they died before or because deemed at too high risk. All of them died of haemorrhage (4,15, 23, 24). Twenty two out of the 41(54%) who had surgery ended with splenectomy (1-3,5,7,8,11-14, 25-36). Seven (15%) were treated with transcatheter arterial embolization. Three with splenic artery aneurysms rupture into the stomach (25, 37, 38) and four with splenic artery aneurysms rupture into the colon (11,39-41) two out of seven failed (29%) and had surgeries (11,25) all for splenic artery pseudoaneurysms. Ten (24%) had ligation of the splenic artery aneurysms with out splenectomy (3,6,9,21,42-44). The mortality in patients presented with splenic artery aneurysm rupture into the stomach is 20%, 37.5% into the colon and 67% into both stomach and colon. Patient with pseudoaneurysm had higher mortality than the one with true splenic artery aneurysm (31% Vs 21%). The overall mortality is 29%. All the patients who had the transcatheter arterial embolization survived (0% mortality). Reviews of the literature by Shanley's on SAAs from 1985 to 1995 showed that as many as 12% of SAAs were reported to be primarily treated by transcatheter arterial embolization (52). A comprehensive review of the English literature by Stabile(53) for acute hemorrhage from pancreatic pseudocysts and ruptured pseudoaneurysms revealed overall mortality of 37%, operative mortality 29% and 12.5% mortality for the one managed by transcatheter arterial embolization. In a review of the literature by Heggtveit (54) for cases of splenic artery aneurysm with intragastric rupture before 1963, six patients out off sixteen under want surgical intervention only one survived which make the surgical mortality for this condition 83% prior to 1963 comber to 29% for the current review.


Conclusion

 

       The present case and the review of the literature demonstrates that in selected patients with appropriate arterial anatomy, transcatheter embolization in experienced hands is both safe and often effective method of management for splenic artery aneurysms ruptured into the gastrointestinal tract. 

 

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