The Isolated Internal Iliac Artery AneurysmA Review

F.P. Dix, M. Titi and H. Al-Khaffaf

Abstract

Background

The isolated internal iliac artery aneurysm (IIIAA) is rare but rupture has a high mortality rate. This paper reviews the available literature regarding the epidemiology, aetiology, natural history, diagnosis and management with a focus on aneurysms of atherosclerotic origin.

Methods

A literature search was performed using internet databases PubMed, Medline and Medscape followed by manual cross referencing of relevant articles. Data were retrieved from the papers, tabulated and analysed to form a review of atherosclerotic IIIAA.

Results

Three hundred and seventy-two papers were found relating to internal iliac artery aneurysms in general and 82 were directly relevant to this paper, reporting 94 cases of atherosclerotic IIIAA. For atherosclerotic aneurysms, the median (range) age was 71.9 (4789) years and 95% were male. The natural history is unclear but is probably one of increasing size, with corresponding increased risk of rupture. Presentation was with rupture in 40%, leading to rapid death if untreated. The death rate in the group as a whole was 31%. The median (range) size of aneurysms at diagnosis was 7.7 (213) cm and death was significantly associated with rupture (Spearman correlation coefficient r=0.327, p=0.007). Symptoms included abdominal pain (31.7%), urological symptoms (28.3%), neurological symptoms (18.3%), groin pain (11.7%), hip or buttock pain (8.3%) and gastrointestinal symptoms (8.3%). Diagnosis may also be coincidental as a result of investigation for other conditions. Of particular use in diagnosis and assessment are ultrasound, computerised tomography and magnetic resonance angiography. Surgical treatment is difficult but can be achieved by ligation, excision or endoanneurysmorrhaphy. More recently, radiological treatments include coil embolisation and endoluminal stenting (often in combination) with the established advantages of endovascular repair have yielded promising short term results, although long term follow-up is required to assess complications and the durability of the devices.

Conclusions

Atherosclerotic IIIAA is a rare condition and if undiagnosed is often fatal. Early diagnosis and treatment may reduce morbidity and mortality particularly with the advent of endovascular techniques.

Article Outline

1. Introduction

2. Methods

2.1. Data sources

2.2. Statistical analysis

3. Results

3.1. Epidemiology

3.2. Aetiology

3.3. Natural history

3.4. Presentationsymptoms and signs

3.5. Asymptomatic

3.6. Rupture

3.7. Urological symptoms

3.8. Neurological manifestations

3.9. Pelvic mass

3.10. Gastrointestinal symptoms

3.11. Other symptoms

3.12. Diagnosis and investigation

3.13. Treatment

3.14. Proximaldistal ligation

3.15. Excision/resection

3.16. Endoanneurysmorrhaphy

3.17. Embolisation

3.18. Endoluminal stenting

4. Conclusions

References

1. Introduction

Isolated internal iliac artery aneurysm (IIIAA) is a rare but serious condition of which vascular surgeons need to be aware. Untreated the natural history is probably one of continued expansion and rupture with a high mortality rate, but with correct and timely treatment survival can be improved. This paper reviews the available published literature to provide a comprehensive review of the epidemiology, aetiology, presentation, diagnosis and management of these aneurysms with particular reference to the commonest form, the atherosclerotic internal iliac aneurysm.

2. Methods

2.1. Data sources

An internet based literature search was performed using the phrases internal iliac artery aneurysm, hypogastric artery aneurysm and endovascular repair of iliac artery aneurysm. The search included PubMed, Medline and Medscape databases to identify all published reports in the English language. Manual cross-referencing from the reference lists of all relevant articles identified further papers included in this review.

2.2. Statistical analysis

Data were retrieved from each relevant paper, entered into a purpose-designed database and analysed using SPSS 11.0 for Windows. Non-parametric tests were used for analysis. Data are presented as medians and ranges and data were compared using the MannWhitney U test and the Spearman correlation coefficient.

3. Results

Of the 372 papers found relating to internal iliac artery aneurysms in general, 82 were directly relevant to this paper and are discussed. Because this is a rare condition the literature abounds with case reports but there are few large scale series or reviews and no randomised trials relating to management.

3.1. Epidemiology

An IIIAA can be defined as a two-fold increase in the size of the artery without a coexisting aneurysm in another location.[1] Iliac artery aneurysms usually present in conjunction with aneurysms of the abdominal aorta where the incidence is approximately 10% but isolated iliac aneurysms are rare and occur in only 2%.[2] An IIIAA is even more unusual with an incidence of 0.4%.[3, 4, 5] and [6] Lucke and Rea (1921) found only one case in a series of 12,000 autopsies in which they also found 321 aneurysms elsewhere.[3] Brunkwall et al. estimated the prevalence to be lower at 0.03% which was also based on a large series of autopsy data.[7].

The majority of patients are elderly males aged 6570 years but presentation from 2692 years has been described, as well as cases in neonates and young parous women. It is much more common in men, with a male to female ratio in most studies of 6:1 or greater.[8, 9, 10] and [11] Table 1 shows the data of all available published reports (from 1947 to the present day) of isolated atherosclerotic internal iliac artery aneurysms (excluding all cases with aneurysms elsewhere and aneurysms of non-atherosclerotic origin). Of the 94 cases reported to date, 63 reported the sex in which 95% (60/63) were male and 5% (3/63) were female. The median (range) age from these published reports (63 reported age) was 71.9 (4789) years.

Table 1. Published reports of atherosclerotic isolated internal iliac artery aneurysms

3.2. Aetiology

The causes of IIIAA are atherosclerosis, infection, trauma and disorders of the arterial wall. The majority of cases are related to progressive atherosclerosis. In the early 20th Century aneurysms caused by syphilis and tuberculosis were described.[12] Other infective causes include Salmonella spp., Staphylococcus aureus, Escherichia coli, Pseudomonas spp. and Klebsiella spp.[13] and [14] Rare iatrogenic cases occur in neonates secondary to umbilical artery catheterisation causing retrograde sepsis.[15] Trauma of many causes is known to cause pseudoaneurysm and this includes aneurysms of the internal iliac artery: penetrating injury to the pelvis and pelvic fracture have both been described.[2, 10, 16, 17] and [18] Iatrogenic trauma during hip replacement, lumbar disc surgery and pelvic surgery has also been described.[18] and [19] An important subgroup of patients are young females who have undergone pregnancy and delivery (in particular a traumatic delivery), forceps delivery or Caesarian section.[9, 12] and [16] Connective tissue disorders and diseases of the arterial wall such as Marfan and Ehlers-Danlos syndrome, fibromuscular dysplasia, Takayasu's arteritis, Kawasaki disease, Behcet's disease, cystic medial necrosis and spontaneous dissection have all been reported as rare causes.[1, 19] and [20]

3.3. Natural history

The natural history of these aneurysms is unclear but probably is one of increasing size, associated with increasing risk of rupture, as is the case with aortoiliac aneurysms. In the study by Richardson and Greenfield, the mean size of IIIAA at diagnosis was 7.7 cm with a 33% incidence of rupture, although the mean size of ruptured aneurysms was 5.6 cm.[2] In a study by Brin and Busuttil, rupture occurred in 38% with a mortality of 58% although they could not find a direct relationship between the size of the aneurysm and the risk of rupture.[9] In the same study conservative management was associated with a high risk of rupture and death. Non-operative management tends to be reserved for patients unfit for surgery or those with small aneurysms but rupture in vessels <3 cm has been described and several authors suggest repair of aneurysms >3 cm as the risk of rupture is 1431%.[2, 8] and [21]

From the data in Table 1, the nature of the aneurysm at diagnosis was reported in 70 cases: of these 40% (28/70) presented with rupture and 60% (42/70) presented without rupture. Of the whole group 69% (49/71) survived and 31% (22/71) died but the data are spread across several decades and more recent treatments have improved mortality. Of those that ruptured and in which the outcome was reported (27 cases), 48% (13/27) survived compared to 52% (14/27) that died. These figures are surprisingly similar to those of Brin and Busuttil.[9]

From the same data in Table 1 the median (range) size of IIIAA was 7.7(213) cm (n=60). The median size of ruptured aneurysms was 7.0(513) cm compared to 6.0(211) cm in the non-ruptured group although this difference was not statistically significant (p=0.274, MannWhitney U test). There appears to be no significant correlation between the size of the aneurysm and the risk of rupture (r=+0.161 Spearman, p=0.279). The survival of the patient was not associated with age (r=+0.071 Spearman, p=0.578) and rupture of the aneurysm was not related to age (r=+0.117 Spearman, p=0.359). The death of the patient was, however, significantly associated with rupture of the aneurysm (r=+0.327 Spearman, p=0.007).

3.4. Presentationsymptoms and signs

An IIIAA may be asymptomatic at diagnosis, present with frank rupture or present with symptoms caused by pressure on adjacent organsthe ureter anteriorly, the internal iliac vein and lumbosacral trunk posteriorly, the external iliac vein and obturator nerve laterally and the colon and small bowel medially.[22] From the data in Table 1, in 34 of the 94 cases described in the literature there was not enough information in the paper to elicit symptoms at presentation. In the remaining 60 cases, 13.3% (8/60) were asymptomatic at diagnosis. Other presentations included abdominal pain in 31.7% (19/60), urinary symptoms or renal failure in 28.3% (17/60), lumbo-sacral pain in 18.3% (11/60), groin pain in 11.7% (7/60), hip or buttock pain in 8.3% (5/60), rectal bleeding or constipation in 8.3% (5/60) and deep vein thrombosis in 1.7% (1/60).

3.5. Asymptomatic

The internal iliac artery lies deep in the pelvis and an aneurysm may remain asymptomatic and unrecognised until it ruptures. Early diagnosis is unusual unless there a high index of suspicion or unless it is diagnosed coincidentally by radiological imaging for other reasons. There is variability in the literature as to the percentage that are thought to be asymptomatic at the time of diagnosis. McCready et al. reported that 78% were asymptomatic.8 Kasulke et al. and Brin and Busuttil reported lower rates at 43 and 29%, respectively.[6] and [9]

3.6. Rupture

Aneurysm rupture may be the first presentation with hypotension, abdominal, groin and thigh pain. Retroperitoneal rupture may be contained but intraperitoneal rupture can lead to rapid death. Rupture into the rectum, ureter, bladder and rectus sheath have all been reported.[23, 24, 25, 26] and [27] When rupture occurs, mortality usually ranges from 50 to 100%.[6, 28] and [29] Mortality is often due to a delay in diagnosis and the difficulties of intra-operative management, with subsequent major haemorrhage.[28] and [30] A diagnosis made before rupture may allow a planned aneurysm repair, which in recent studies has a much lower mortality rate of less than 10%.[28]

3.7. Urological symptoms

Urological symptoms are common and reported to occur in 54% of cases.[31] The first published report described a man with pulsatile micturiton and obstructive urinary symptoms due to compression of the bladder by the aneurysm.[32] Difficulty in micturition and urinary retention are commonly described.[4] and [33] Ureteric colic, hydronephrosis, pyelonephritis and renal failure can occur secondary to ureteric obstruction.[6, 34] and[ 35] Intermittent haematuria, rupture into the bladder, ureter and scrotal ecchymosis have all been described.[6, 26, 36 ] and [37] Ureteric stenting prior to elective repair may improve renal failure and aid identification of the ureter at the time of surgery but cystoscopy has been reported to cause massive haematuria.[38] and [39] An expanding aneurysm displaces the ureter antero-laterally making it densely adherent and prone to injury at the time of surgery.[40]

3.8. Neurological manifestations

Ipsilateral leg pain with neurological signs have been described in both the elective and emergency setting.[4] Psoas irritation, paresis, sciatic neuralgia and lumbosacral pain have been reported by several authors.[4, 10, 26] and [34] Compression of the pelvic and lumbosacral nerve roots is reported to occur in 1015% of patients.[38, 41, 42] and [43] Krupski et al. reported a case of obturator neuralgia and Ijaz and Geroulakos reported a case in which an the aneurysm mimicked a hip fracture.[19] and [26]

3.9. Pelvic mass

A tender, palpable mass in the iliac fossa is found in approximately 55% of cases.[2] and[ 9] It should be pulsatile, distinguishing it from other causes of mass such as appendiceal mass, hernia and colonic tumour.[4] On rectal or vaginal examination a mass can often be palpated in the pelvis and many authors regard this as an important part of the clinical examination.[4] and [10]

3.10. Gastrointestinal symptoms

Rectal bleeding is usually associated with rupture of the aneurysm into the colon.[34] Compression or deviation of the colon by the aneurysm may cause constipation, tenesmus and rectal pain.[6, 24] and [44] Wu et al. reported a case of intermittent rectal bleeding, anaemia and a rectal mass confirming that rectal examination is important in diagnosis.[22] Major rectal bleeding secondary to ilio-rectal fistula has also been reported.[45] A colostomy may be required to relieve colonic obstruction caused by compression from the aneurysm and retroperitoneal haematoma or where the aneurysm has ruptured in to the colon.[46]

3.11. Other symptoms

Obstruction to ilio-femoral venous flow causing oedema of the lower limb, deep vein thrombosis and pulmonary embolism have all been reported.[6, 10, 47, 48] and [49] Heart failure from an arterio-venous communication has been reported.[50]

3.12. Diagnosis and investigation

Abdominal and pelvic plain films may reveal the curvilinear calcified wall of an aneurysm in the lower quadrant but are unhelpful in assessing the extent or size.[8] and [10] Initial investigation involved aortography but this has been superceded by more modern investigations.[4] Femoral angiography was also popular and was easier to perform, particularly on the operating table and could also identify an arterio-venous fistula.[2, 4] and [22] Intravenous pyelography may show a hydronephrosis and hydroureter or a poorly functioning kidney with displacement of the ureter by a mass lesion.[26, 34] and [35] Cystoscopy may reveal a pulsatile pelvic mass with extrinsic compression of the bladder.[34]

Colour flow Doppler studies are useful in establishing the initial differential diagnosis of other masses of the pelvis and can demonstrate pulsatile flow within the aneurysm.[51] and [52] Abdominal and pelvic ultrasound imaging can easily and non-invasively show the aneurysm but are limited by operator ability and the deep location of the iliac artery in the pelvis, overlying bowel gas and the tortuous nature of the artery.[34, 38] and [51] Ultrasound is also useful in revealing renal cortical thinning due to chronic ureteric obstruction caused by the aneurysm.[35]

Computerised tomography (CT) is now well established in the diagnosis of most intra-abdominal aneurysms.[53] CT with contrast demonstrates the aneurysm site, size and relationship to other organs and is now routinely recommended for elective repair.[8] Contrast enhancement may demonstrate retroperitoneal haemorrhage and displacement of other structures.[2, 19] and [50] The more widespread use of CT for other abdomino-pelvic pathology is increasing the coincidental identification of more aneurysms at an earlier stage.

Magnetic resonance (MR) imaging can also demonstrate the aneurysm and its anatomical relationship to other structures. MR angiography is now able to produce excellent quality images of the vascular tree aiding diagnosis and planning treatment.

3.13. Treatment

The current available treatments for IIIAA are one or a combination of ligation, excision, endoanneurysmorrhaphy, embolisation and endoluminal stenting. Any of these procedures may also require extra-anatomic bypass to maintain flow to the pelvis or limb. Conservative treatment is associated with continued expansion and operative mortality in the emergency rupture setting is much higher than in the elective setting suggesting a benefit of early aggressive treatment of these aneurysms.[9, 28] and [54] Overall peri-operative mortality remains high at 3350% in emergency operations but has reduced to 711% for elective procedures.[2] In the elective situation, surgery can be planned after appropriate investigation and performed by experienced surgeons familiar with the anatomy of the pelvic vessels and potential pitfalls of this surgery.[55] In the emergency situation there may be no time for investigations and the available surgeon may be less experienced. Levi and Schroeder described repair of four IIIAA in which median blood loss was 4700 ml, median transfusion requirement was 11 units per operation and there were no peri-operative deaths.[56] There may be a case for surgery to be performed in vascular units with use of a cell saver.

Unilateral ligation is well tolerated if the contralateral artery is free of disease. When there are bilateral internal iliac aneurysms, the inferior mesenteric artery should be evaluated for obvious atheromatous disease. Ischaemic damage to the pelvic viscera can ensue when the internal iliac aneurysm is excluded producing ischaemia of the colon and bladder and causing vasculogenic impotence.[57] and [58] If proximal occlusion of the inferior mesenteric artery exists, an attempt should be made to reimplant it; if not bypass is recommended.[30]

3.14. Proximaldistal ligation

Historically, surgery involved proximal ligation of the aneurysm.[5, 24, 32, 34] and [59] MacLaren performed the first successful ligation of an IIIAA in a woman having had a difficult and traumatic instrumental delivery.[59] The advantage of proximal ligation alone is limited dissection in the pelvis reducing the risk of bleeding. However, the aneurysm may refill by retrograde flow with a risk of late rupture and this technique fails to deal with any compressive symptoms.[9, 21, 50] and [60] Proximal and distal ligation in combination are an effective treatment with less risk of refilling but more risk of operative bleeding and ideally all tributaries leading into the aneurysm should also be ligated.[30] Exclusion of the aneurysm (ligation of the proximal part, leaving the distal part alone) seems to be effective in small, localised proximal IIIAA. However, in those extending deeply into the pelvis exclusion appears to be ineffective.[61] Long-term CT surveillance is recommended by these authors where the aneurysm does not decrease in size post-operatively.

From the data in Table 1, 27 patients had proximaldistal ligation making it the most commonly performed procedure although the use of endovascular techniques may become the mainstay of treatment in the future. Of the 25 where the outcome was stated 28% (7/25) died.

3.15. Excision/resection

Many of the early descriptions of surgical treatment involved excision of the entire aneurysm but major bleeding made worse by venous congestion due obstructed pelvic veins led to a high mortality rate.[4, 9] and [32] Several authors do not recommended excision in the emergency situation due to the risk of haemorrhage or damage to nearby structures, particularly the ureter which is often adherent to the wall of the aneurysm secondary to peri-aneurysmal fibrosis.[11] and [62] Achieving repair without complete resection of the back wall of the artery, can prevent massive bleeding.[55] Of the cases in Table 1, 15 had aneurysm resection and of these 26.7% died (4/15).

3.16. Endoanneurysmorrhaphy

Obliterative endoanneurysmorrhaphy has been described in which the sac is opened and the ostial branches oversewn.[5] and [6] This is only suitable for unilateral IIIAA because bilateral endoanneurysmorrhaphy can be associated with buttock necrosis, colitis and paralysis.[63] and [64] Graft interposition is often employed to establish continuity of flow to the pelvis particularly where there are bilateral aneurysms and should at least be attempted on one side. The surgery is often difficult due to the large size of the aneurysm, venous obstruction, difficult access to the distal vessels and retroperitoneal haematoma. Distal vessels passing through the sciatic foramina may cause extensive bleeding if not secured but can be controlled by balloon catheters.[10] Buttock necrosis was associated with a very poor outcome with mortality reported at 100% in the study by Bao.[63] Younger adults may tolerate bilateral aneurysm ligation but the risk is still very high.[65] Of the cases in Table 1, 15 had aneurysmorrhaphy and all survived although this is not necessarily significant.

3.17. Embolisation

Less invasive techniques for the treatment of IIIAA became available in the early 1980s. Embolisation using Gianturco coils (Cook) was described by Perdue in 1983.[10] Arteriography 3 months later confirmed complete obliteration of the aneurysm. Hollis et al. in 1994 described percutaneous embolisation in conjunction with an extra-anatomic bypass.[66] This treatment was particularly useful in patients unfit for open surgery and avoids the potential complications of distal embolisation.[67] Many other authors have reported successful treatment using transfemoral endovascular techniques with limited local trauma, rapid recovery, low blood loss, less anaesthetic requirement and reduced in-hospital stay.[68, 69] and [70] In a study by Kritpracha et al. in which 20 patients underwent coil embolization (12 distally), four became significantly limited by symptoms of claudication from which the authors now recommend that coil placement should be as proximal as possible to prevent interference with pelvic collateral circulation.[71] The risks of distal embolisation can be minimised by using soft-tipped wires and catheters and reducing manipulation.[68] Recognised complications do however, include buttock claudication in 1255% and erectile dysfunction in 113%.[72] Further developments in these techniques have led to a study of embolisation using fibrin sealants.[73] The majority of endovascular treatments for IIIAA now involve a combination of coil embolisation and stenting as discussed below and in the data in Table 1, only three cases had coil embolisation alone.

3.18. Endoluminal stenting

Unlike aorto-iliac aneurysms, at the present time stent grafts cannot be placed directly into the internal iliac artery but internal iliac aneurysms can be excluded by stenting of the external and common iliac arteries. Stenting is regarded as the equivalent to proximal ligation and coiling equivalent to distal ligation.[74] Early techniques involved surgical access to enable deployment of the stent.[75, 76] and [77] Further advances in technology have enabled the use of a truly percutaneous technique.[74] and [78] Razavi et al. performed elective percutaneous endoluminal stenting of iliac aneurysms (not necessarily IIIAA) in eight patients under local anaesthetic.[78] Placement of the stent graft with thrombosis of the aneurysm was successful in all patients. Endoleaks were corrected by further stent placement or coil embolisation. There were no distal emboli, iliac artery occlusions, infections, ruptures or haematomas. This technique offers the advantages of immediate isolation of the aneurysm without the need for general anesthesia, reduced blood loss and reduced hospital stay. The main disadvantage is that any pressure effect symptoms caused by the aneurysm are not relieved by this method, as is the case with simple ligation. Cynamon et al. performed successful coil embolisation in combination with stenting to occlude an internal iliac artery aneurysm.[76] Henry et al. also described coil embolisation of an internal iliac aneurysm in combination with balloon expandable stenting of the external iliac artery.[74] Computerised tomography at 6 months follow-up confirmed complete occlusion of the aneurysm. The same authors recommend that where the aneurysm has a small orifice the technique can be used successfully but where the orifice is large, a covered stent should be used.

Marin et al. used transluminally placed endovascular grafts of PTFE sewn to a balloon expandable stent to occlude various types of iliac artery aneurysms in 11 patients. Grafts were successfully placed in all which included two internal iliac aneurysms.[75] Cormier et al. also recommend the use of covered stents for the endovascular treatment of iliac aneurysms.[79] In their retrospective study, 15 out of 18 patients with internal iliac artery aneurysms (not necessarily isolated internal iliac aneurysms) had exclusion with Gianturco coils 2448 h before stent graft placement. In the remaining three, prior embolisation was not performed for reasons of emergency treatment or high grade ostial stensosis. Parsons et al. placed stent grafts in 28 iliac artery aneurysms some of which were IIIAA using PTFE and balloon expandable stents and achieved an 86% 3-year survival.[80]

There are now case reports of combined embolisation and stenting in patients with ruptured IIIAA.[81] Ricci et al. successfully treated a ruptured internal iliac aneurysm in an 81-year-old man with multiple comorbidities with multiple coils and a covered stent. He was well at 12 months follow-up.[81] Although the use of endovascular techniques is very promising and likely to be used in the treatment of a significant proportion of IIIAA in the future, long term follow-up of both techniques is required to fully establish their complication rate, durability, and patency.

4. Conclusions

Internal iliac artery aneurysms are rare but are a challenge to the vascular surgeon in diagnosis and treatment. Historically, mortality was high particularly in the ruptured case, but this is now improving with earlier diagnosis, better preoperative investigation, better surgery and the advent of endovascular treatments.[82] Prompt diagnosis followed by early treatment is recommended in order to reduce morbidity and mortality.

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