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Interact CardioVasc Thorac Surg 2009;8:384-386. doi:10.1510/icvts.2008.196121 © 2009 European Association of Cardio-Thoracic Surgery
Unusually giant splenic artery and vein aneurysm with arteriovenous fistula with hypersplenism in a nulliparous womanDepartment of C.T.V.S., S.M.S. Medical College and Hospital, Jaipur, Rajasthan, India Received 11 October 2008; received in revised form 28 November 2008; accepted 3 December 2008
*Corresponding author. Room No. 170, R.D. Hostel, S.M.S. Hospital Campus, Jaipur, Rajasthan-302003. Tel.: +919214029283; fax: +141 2619171.
Although splenic artery aneurysm (SAA) is the commonest visceral and third most common intra abdominal aneurysm after aorta and iliac artery, aneurysm of splenic artery along with aneurysm of splenic vein with arteriovenous (a–v) fistula communication between them is a rare entity. Most are <3 cm in diameter. Giant true SAAs are rare and very few lesions >10 cm have been reported. We hereby report a case of an 18 cmx15 cm size splenic artery and vein aneurysm with a–v fistula in an adult female nulliparous woman who presented with progressively enlarging pulsatile mass in the left upper abdomen with long-standing intractable pancytopenia and splenomegaly. Diagnosis was established by CT (computed tomogram) angiogram and laboratory tests. Laparotomy demonstrated huge well-defined aneurysm of splenic artery and vein with splenic a–v fistula, extending in all except the right lower and inferior quadrants of the abdomen along with splenomegaly. Aneurysmectomy with splenectomy was done.
Key Words: Aneurysm; Splenic artery; Intractable pancytopenia; Pulsatile abdominal lump
Splenic artery and vein aneurysm with splenic arteriovenous fistula (SAVF) is rare. It could be congenital in origin, postoperative, post-traumatic, or following mycotic infection [1]. Splenic artery aneurysms (SAAs) are rare findings, are usually single and small lesions, and their size does not usually exceed 3 cm. In most instances, elimination of aneurysm with the SAVF is recommended, even in asymptomatic patients, to prevent complications. A combination of standard transabdominal surgery with transcatheter embolization can be done in selected cases when transcatheter embolization alone is not possible [2]. We herein report an intriguing case of a huge SAA of 18 cm size associated with SAVF with features of hypersplenism in which surgical treatment was required.
A 35-year-old female patient was admitted with chief complaints of dull aching pain and progressively enlarging mass in the left upper abdomen. On examination she had pallor. Huge pulsatile mass was present in the left upper abdomen. Moderate splenomegaly was present and the pulsatile mass was inseparable from the spleen. Laboratory examination showed anaemia (red cell count: 3.27x106/µl, reference range: 4.40–5.60x106/µl; haemoglobin: 6.4 g/dl, reference range: 13.0–16.5 g/dl), leucopenia (wbc count 3x103/µl, reference range: 4–11x 103/µl) with predominant neutropenia and thrombocytopenia (platelet count: 0.5x105/µl, reference range: 1–4x105/µl. Serum renal and liver function tests were normal. An abdominal ultrasound with colour-Doppler examination showed large hypoechoic lesion of size 18 cm in close association to the splenic artery with accelerated blood flow in it with no thrombosis with splenomegaly. Upper GI Endoscopy was unremarkable. Subsequently, an enhanced spiral computed tomography revealed a well-defined rounded 18x15 cm hypodense mass situated anterior and superior to the pancreas and displacing the stomach upward extending up to the splenic hilum. CT angiography confirmed the diagnosis and it also delineated splenic arteriovenous fistula communication (Fig. 1).
Surgical exploration of the abdomen showed a well-defined aneurysmal sac with thin and thick walls and difference in colours demarking venous and arterial portion of the aneurysm with visible flow of blood through venous portion (Fig. 2). The flow inside the sac ceased on compressing the splenic artery at the superior border of the pancreas.
Aneurysmectomy with splenectomy was done using left thoracoabdominal approach.
SAAs constitute 60% of all splanchnic aneurysms [3]. SAAs are second in frequency to aorto-iliac artery aneurysms. The majority of SAAs are <2 cm. Only a few cases with SAA size >10 cm are reported. In the group of patients having a rupture of the aneurysm, an SAA has an average size of 5.5 cm. An aneurysm of 18 cm, as in our case, raised serious concerns regarding the risk of rupture and bleeding. No reference could be traced in the literature to support our view about non-rupture of such a large aneurysm, which was the presence of a–v fistula and insignificant portal hypertension allowing it to drain in relatively low-pressure venous system. In women, pregnancy is a major risk factor for rupture; approximately 95% of them occur in the second trimester [4]. In the majority of patients (74–87%) the SAAs were located in the distal third of the splenic artery, in 22% they were in the middle third and, in the remaining, they were in the proximal third [5–7]. Splenic arteriovenous fistulae are commonly due to spontaneous rupture of a preexisting splenic artery aneurysm into the splenic vein. They are considered either congenital or acquired in origin, and mostly occur in women of childbearing age with a history of multiparity. But our patient was nulliparous. Post-traumatic arteriovenous fistulae usually follow blunt trauma, and normally occur within one year of the trauma [4]. Our patient was nulliparous, had never been pregnant and there was no history of abdominal trauma. Patients suffering from splenic arteriovenous fistulae are characterized by the effect of portal hypertension such as splenomegally, barometric portal hypertension, esophageal varices, gastrointestinal bleeding, ascites, and diarrhoea are observed. The most frequently observed symptoms (61%) were machinery-type bruit over the left flank. A small number of patients (16%) had no symptom of portal hypertension as in our case. Abdominal ultrasound with Doppler is an important modality for physicians, and enables the investigator to document portal hypotension and splenomegally, rule out advanced chronic liver disease, and document the presence of splenic arteriovenous malformation. While most reports have established the diagnosis through abdominal arterial angiography, recent reports have stressed the possibility and role of Doppler ultrasound representing a fast and non-invasive strategy. In patients where arteriovenous fistula has been suspected clinically, Doppler ultrasound should be the first diagnostic study performed, as is demonstrated in the present case report. Contrast enhanced CT-scans and CT angiogram is important in the evaluation of this condition. Once the diagnosis has been confirmed, a therapeutic strategy needs to be determined. In principle, one of two options exist: (1) Operative treatment with dissection of the SAA and fistula, and frequently splenectomy, being the more invasive. (2) Percutaneous catheter embolization. The current data support the use of endovascular procedures as first choice of treatment for SAA with SAVF. A consideration to preserve the spleen and its function should be made in all cases, though splenectomy may be necessary in suspected cases of associated hypersplenism as was the case in our patient. Open surgery is reserved mostly for the treatment of complications or if endovascular procedures fail, or are not possible, or when patients with chronic renal insufficiency need urgent intervention. The location on the artery of the aneurysm makes a serious difference in surgical management. If the aneurysm is located in the distal third of the artery and the aneurysm has to be resected, the patient may ultimately require a splenectomy. If the aneurysm is located in the proximal third of the artery, splenic conservation should be attempted [8]. In our patient it was located in the middle and distal portion of the splenic artery where operative technique was adopted to deal with such a large aneurysm of 18 cmx15 cm and to perform splenectomy in the same setting for hypersplenism.
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Abstract
1. Introduction
