Iliac vein aneurysms are extremely rare, even amongst vein aneurysms. We discuss the case of a 26-year-old man with an external iliac vein aneurysm, likely secondary to iatrogenic vascular trauma in the neonatal period. It is the ﬁrst reported case of an iliac vein aneurysm presenting with lower urinary tract symptoms. Attempts at endovenous management were unsuccessful and therefore the patient underwent open aneurysmectomy. A PubMed literature search revealed a total of nine case reports of iliac vein aneurysms published in English since 2011. We discuss the aetiology, presentation, investiga- tion and management of iliac vein aneurysms and compare to our own case. subsequently underwent venography, which conﬁrmed an INTRODUCTION aneurysmal EIV, with massive collateral dilatation of trans- Iliac vein aneurysms are extremely rare, even amongst vein pelvic veins and complete occlusion of the right common fem- aneurysms. We discuss the case of an external iliac vein (EIV) oral vein (CFV). A concurrent attempt at endovenous stenting aneurysm, likely secondary to iatrogenic vascular trauma in was made, but was unsuccessful as the CFV could not be the neonatal period. passed with a guide-wire and no alternate route was found possible. Further characterization by CT demonstrated a dilated and tortuous right internal iliac vein (IIV) feeding the aneurysm CASE REPORT (Fig. 1). A multidisciplinary decision was made to proceed to A 26-year-old man was referred to our vascular surgery tertiary operative repair, in light of the patient’s debilitating urinary fre- referral service from his local hospital with leg size discrepancy quency from the mass effect of the aneurysm. Pre-intervention (right > left) and prominent right leg varicose veins distally. He venous severity scoring was not pursued as the patient’s symp- had lower urinary tract symptoms (frequency >30 times per toms were only bladder-related. The patient underwent an day) for a year prior to presentation but had no leg symptoms open aneurysmectomy via a Rutherford-Morrison incision, in relation to his varicosities. His past medical history was of with concurrent right retrograde ureteric stent placement. premature birth, as one of triplets, who spent a prolonged peri- Intra-operatively, a wide-necked aneurysm was conﬁrmed to od on a neonatal intensive care unit. The patient thought he arise from the EIV (Fig. 2) in a saccular fashion. The aneurysm may have had a deep vein thrombosis in his right leg as a child. was clamped at the neck and closed with 3-0 Prolene (Ethicon, An MRI of his right thigh and pelvis demonstrated a 5 cm ven- Inc., New Jersey, USA) leaving a normal calibre EIV (Figs 3 and 4). ous aneurysm (EIV), indenting the bladder, and generalized dis- The IIV remained patent at the end of the procedure and a tal venous dilatation of both deep and superﬁcial veins. He venous bypass was not performed. The patient recovered well Received: March 21, 2018. Accepted: May 18, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact firstname.lastname@example.org Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy115/5020654 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 A. E. Fanshawe et al. Figure 1: CT scan demonstrating right iliac vein aneurysm (5.7 cm) (red arrow) compressing the patient’s full bladder. Figure 4: Intra-operative appearance post aneurysmectomy. Figure 2: Intra-operative appearance of the external iliac vein aneurysm. A retroperitoneal approach was used via a Rutherford-Morrison incision. Laterally, the external iliac artery is controlled with a vessel loop. Figure 5: Post-operative CT scan demonstrating no evidence of residual aneur- ysm, or bladder compression. A residual dilated and tortuous collateral vessel remains communicating between the right internal iliac vein and right com- mon femoral vein (red arrow). post-operatively and was discharged home four days later, with removal of the ureteric stent prior to discharge. He has been fol- lowed up 6-monthly since and remains well at 2 years’ post- operatively. CT venogram has shown a good post-operative result, with no residual aneurysm (Fig. 5). The patient’s pre- operative lower urinary tract symptoms have improved. He did not develop any leg symptoms. DISCUSSION Vein aneurysms may be deﬁned as solitary venous dilatations that communicate with a main venous structure, without con- temporaneous arteriovenous ﬁstulae . Iliac vein aneurysms are extremely rare, even amongst vein aneurysms. Approximately 40 iliac territory venous aneurysms have been described in the existing literature. Zou et al.  performed a review of these up to 2011. We present a review of nine new cases in the literature that Figure 3: Clamped external iliac vein aneurysm. have been published since 2011 (Table 1). A systematic PubMed Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy115/5020654 by Ed 'DeepDyve' Gillespie user on 21 June 2018 External iliac vein aneurysm 3 Table 1 The demographics, presentation, nature, location, size, imaging, management and outcome of iliac vein aneurysms reported in English since 2011 (PubMed search) Author Age and Symptoms Associated anomalies Primary or Location Size Imaging Management Outcome gender secondary? Fanshawe 26M Lower-urinary tract symptoms DVT as a child Secondary Right EIV 4 cm CT, MRI and venography Open aneurysmectomy with Well, at 24 months post-op. et al. 2017 primary closure CT conﬁrming no residual (Current case) aneurysm. Resolving lower urinary tract symptoms. Audu et al.  63M Left testicle and groin pain Lower extremity varicose Primary Left IIV 3.1 × 2.2 cm MRI and venography Endovenous embolisation Symptom resolution at 1- veins month follow-up, with CT conﬁrming aneurysmal occlusion. Park et al.  63F Acute severe abdominal pain None Primary Right EIV 4×5cm CT Open aneurysmectomy with Well, at months post-op, primary closure with Duplex USS every 6 months. Shah et al.  22F Incidental (found during work- Patent foramen ovale; IVC Primary Right EIV Not stated CT Open aneurysmorrhaphy Asymptomatic at 4-year up of bilateral acute limb obstruction; bilateral with primary closure follow-up, with ischaemia—treated with proximal common iliac surveillance Duplex USS bilateral femoral stenoses demonstrating no embolectomies) recurrence. Banzic et al.  24F Non-healing skin ulceration Misdiagnosis of Klippel– Primary Left CIV 4 cm CT, prior Duplex USS Endovenous option Diagnosis of Parkes-Weber below left knee Trenaunay syndrome as a presented; patient syndome (multiple AV child; Right leg declined treatment. ﬁstulas throughout the lengthening left limb); Well at 2 years post diagnosis. Escobar et al.  54F Incidental (found during None Not stated Right EIV Not stated Not performed Not stated Not stated. laparoscopic retroperitoneal dissection for endometrial adenocarcinoma) Hosaka et al.  22F Sudden-onset dyspnoea (PE) None Primary Right EIV 3.7 cm CT Open aneurysmectomy with Well, with patent EIV at 8- great saphenous venous month follow-up. patch graft Todorov et al.  62M Incidental (elevated PSA) Persistent left leg swelling Secondary Left EIV 8 × 5×6cm CT, MRI and ﬂuoroscopic Endovenous stent graft Stent graft patent, no leak following ankle fracture; venogram and IVUS and no migration. Post-traumatic superﬁcial intra-operatively Resolution of left leg femoral AV ﬁstula of swelling. Annual follow- same leg (ligated years up with Duplex. previously) Javaraj et al.  37F Left gluteal pain None Primary Left EIV 3.6 cm Duplex USS, CT and Hybrid repair with open Post-op venography venography aneurysmectomy and showing no residual primary closure over aneurysm. Well, at 4 angioplasty balloon as a months post-op. mandrel Zou et al.  14F Sudden-onset dyspnoea and None Primary Left EIV Not stated Duplex USS, CT Mechanical fragmentation Asymptomatic at 16 syncope (PE) and low-molecular months. weight anticoagulant for PE. Surgery offered; family declined further treatment. Abbreviations: M = male; F = female; DVT = deep vein thrombosis; EIV = external iliac vein; CT = computed tomography; MRI = magnetic resonance imaging; post-op = post-operative; IIV = internal iliac vein; CIV = common iliac vein; IVC = inferior vena cava; USS = ultrasound scan; AV = aterio-venous; PE = pulmonary embolus; PSA = prostate speciﬁc antigen; IVUS = intravascular ultrasound. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy115/5020654 by Ed 'DeepDyve' Gillespie user on 21 June 2018 4 A. E. Fanshawe et al. search was performed to identify these, using the search terms: CONFLICT OF INTEREST STATEMENT iliac; vein; venous; aneurysm; external; internal in various combi- None declared. nations. All relevant articles written in English were reviewed. Iliac vein aneurysms may be primary or secondary, in asso- ciation with previous trauma or vascular malformation syn- REFERENCES dromes, e.g. Parkes-Weber . In this case, the iliac vein 1. Audu CO, Boniakowski AE, Robinson S, Dasika NL, aneurysm was secondary and most likely developed as a result Wakeﬁeld T, Coleman DM. Internal iliac venous aneurysm of iatrogenic vascular trauma (multiple femoral central venous associated with pelvic venous insufﬁciency. J Vasc Surg catheters) in the neonatal period. Iliac vein aneurysms can be Venous Lymphat Disord 2017;5:257–60. difﬁcult to diagnose and the differential may include groin her- 2. Zou J, Yang H, Ma H, Wang S, Zhang X. Pulmonary embol- nias or lymphadenopathy. They are often asymptomatic, but ism caused by a thrombosed external iliac venous aneur- may present with thomboembolism or, rarely, abdominal pain. ysm. Ann Vasc Surg 2011;25:982.e15–8. This is the ﬁrst reported case of an iliac vein aneurysm present- 3. Abbott OA, Leigh TF. Aneurysmal dilatations of the superior ing with lower urinary tract symptoms. Investigations of choice vena caval system. Ann Surg 1964;159:858–72. include CT (venous phase), real-time venography and MRI. All 4. Park JS, Kim JY, Kim M, Park SC, Lee KY, Won YS. Ruptured three were used in the case presented. aneurysm of the external iliac vein. J Vasc Surg Venous Complications associated with iliac vein aneurysms include Lymphat Disord 2016;4:92–4. rupture  and thromboembolism. Primarily to prevent 5. Hosaka A, Miyata T, Hoshina K, Okamoto H, Shigematsu K. thromboembolism, operative management has been hitherto Surgical management of a primary external iliac venous recommended. Operative strategies include excision, with or aneurysm causing pulmonary thromboembolism: report of without interposition grafting, or tangential excision—as in the a case. Surg Today 2014;44:1771–3. case we present here—with or without patch repair, if residual 6. Todorov M, Hernandez D. Endovascular exclusion of a large vessel calibre or wall fragility is a concern. Hosaka et al.  used external iliac vein aneurysm. J Vasc Surg Venous Lymphat a dual saphenous vein patch in their operative technique. Disord 2013;1:301–3. Endovenous strategies of both stent grafting and embolisation 7. Jayaraj A, Meissner M. Novel repair of an external iliac vein have also been employed with success [1, 6]. Javaraj et al. aneurysm. Ann Vasc Surg 2012;26:859.e13–5. also performed a successful hybrid procedure in 2012. 8. Shah SK, Shah KB, Clair DG. External iliac vein aneurysm as The rarity of vein aneurysms, together with the heterogen- a cause of paradoxical embolism. J Vasc Surg Venous eity of presentation, investigation and management make it Lymphat Disord 2015;3:322–4. difﬁcult to draw meaningful conclusions regarding their opti- 9. Banzic I, Brankovic M, Koncar I, Ilic N, Davidovic L. Unusual mal treatment. This case is the ﬁrst reported of an EIV aneur- case of Parkes Weber syndrome with aneurysm of the left ysm presenting with lower urinary tract symptoms, due to a common iliac vein and thrombus in inferior vena cava. Ann compressive effect on the bladder. Vasc Surg 2015;29:1450.e17–9. 10. Escobar PF, Salgueiro-Bravo JM, Del Pilar Rodriguez LG, ACKNOWLEDGEMENTS Vaillant A. External iliac vein aneurysm. J Minim Invasive None declared. Gynecol 2015;22:1132. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy115/5020654 by Ed 'DeepDyve' Gillespie user on 21 June 2018
Journal of Surgical Case Reports – Oxford University Press
Published: May 29, 2018
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