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REVIEW

The Persistent Sciatic Artery

I.M. van Hooft

a

, C.J. Zeebregts

b

, S.M.M. van Sterkenburg

a

, W.R. de Vries

a

,

M.M.P.J. Reijnen

a,

*

a

Alysis Zorggroep, Location Rijnstate, Department of Surgery, Division of Vascular Surgery, Wagnerlaan 55, 6815 AD Arnhem, The Netherlands

b

University Medical Center Groningen, Department of Surgery, Division of Vascular Surgery, Groningen, The Netherlands Submitted 2 December 2008; accepted 26 January 2009

Available online 20 February 2009

KEYWORDS

Anomaly; Complication;

Persistent sciatic artery; Review

Abstract Background: A persistent sciatic artery (PSA) is a rare vascular anomaly with an estimated incidence of 0.03e0.06%. During early embryonic development, the sciatic artery usually disappears when the superficial femoral artery has developed properly. This study aimed to assess the clinical presentation and outcome of a PSA.

Method: A systematic review of all cases of PSA published between 1964 and 2007 was per-formed.

Results: In this review, 159 PSAs were described in 122 patients. The mean age at which the PSA was discovered was 57 years, and the incidence was equally distributed with regards to gender. The majority of PSAs was unilateral (70%) and of the complete type (79%). Ninety-one patients (80%) presented with symptoms including intermittent claudication, ischaemia, a pulsating mass or neurological symptoms. An aneurysm was found in 48%, a stenosis in 7%, an occlusion of the PSA in 9% and an occlusion of an artery distal to the PSA in 6% of the subjects. The treatment depended on the symptoms and classification of the PSA. In nine cases (8%), an amputation was required eventually.

Conclusion: The PSA is a rare anomaly with a high incidence of complications including aneu-rysm formation and ischaemia that may lead to amputation. Strategies for follow-up could not be deduced from the available literature.

ª 2009 European Society for Vascular Surgery. Published by Elsevier Ltd. All rights reserved.

In 1832, Green1first reported the presence of a persistent sciatic artery (PSA) in a post-mortem case. The sciatic artery is a branch of the umbilical artery and the principal

arterial supply of the developing lower limb during the 6-mm stage of embryonic development. During the early embryonic phase, the sciatic artery supplies blood to the developing lower limb,2 while in the 12-mm stage it develops into a common and a superficial femoral artery (SFA).2Thereafter, the superficial artery descends to knee level and terminates at the bifurcation of the medial

* Corresponding author. Tel.: þ31 263786908; fax: þ31 263786627.

E-mail address:mmpj.reijnen@gmail.com(M.M.P.J. Reijnen).

1078-5884/$34ª 2009 European Society for Vascular Surgery. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ejvs.2009.01.014

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descending geniculate artery and the superior communi-cating branch. The latter joins the sciatic artery just above the knee level. In the 3rd month of embryonic develop-ment, the sciatic artery is normally completely interrupted and the blood supply to the lower limb is completely taken over by the SFA.3,4 Meanwhile, the umbilical artery

develops into the internal iliac artery. At that time, a part of the sciatic artery involutes and other parts develop into permanent arteries: the proximal part of the sciatic artery gives rise to the inferior and superior gluteal arteries2and

the distal part of the sciatic artery forms the peroneal and popliteal arteries. However, in some post-mortem cases,5,6 the PSA and the internal iliac artery have been shown to have existed simultaneously. Some authors, therefore, assume that the internal pudendal artery is a branch of the internal iliac artery, instead of being a remnant of the PSA.5e7

Several authors have stated that the sciatic artery may persist when the femoral artery develops incompletely.8 The reasons for an incomplete or absent development of the femoral artery are unknown. When the sciatic artery persists, it is usually a tortuous and enlarged vessel.9

Commonly, the internal iliac artery has a larger diameter than the external iliac artery, supplying blood to the lower leg through the PSA.2 The anatomical position of a PSA differs from the position of the femoral arteries. The sciatic artery starts at the internal iliac artery and runs through the greater sciatic foramen, from where its course is close to the sciatic nerve. In some cases, the sciatic artery actually lies inside the sciatic nerve sheath.10Distally, the artery lies deep in the major gluteal muscle and follows a course along the adductor magnus muscle. Finally, the sciatic artery reaches the popliteal artery running through the popliteal fossa, lateral to the insertion of the adductor magnus muscle.10

Pillet et al. have described four different types of a PSA11,12 (Fig. 1): type 1 represents a complete PSA in combination with a normal femoral artery; in type 2, a complete PSA is present in combination with an incom-pletely developed femoral artery e in subtype 2a the SFA is present, but does not reach the popliteal artery (Fig. 2), while in subtype 2b the SFA is absent; type 3 represents an incompletely PSA, in which only the upper part has per-sisted and the femoral arteries are normally developed and in type 4, an incomplete PSA is found, in which only the lower part has persisted with normally developed femoral arteries. Gauffre et al.13 added a fifth type: a PSA

originating from the median sacral artery. Two subtypes have been described: type 5a with a developed SFA and type 5b with an undeveloped superficial artery. Bower et al.14 have proposed another classification system that differentiated a complete from an incomplete type. The PSA is considered complete when it is the main blood provider to the lower limb. In this type, the superficial artery is mostly hypoplastic and ends in the thigh. The PSA is considered incomplete when the SFA is the main blood provider to the popliteal artery. In this type, the sciatic artery is usually hypoplastic and terminates in the thigh.

We conducted a systematic review of all cases described in literature between 1964 and 2007. The classification, symptoms, diagnostic evaluation, complications and treat-ment options of the PSA are all discussed in this article.

Method

A literature search identified articles focussing on the key issue ‘sciatic artery’ using the Cochrane and PubMed databases. The articles were selected based on their title

Type1 Type 2a Type 2b Type 3 Type 4 Type 5a Type 5b

Figure 1 Classification system according to Pillet et al. (modified by Gauffre et al.).

Figure 2 a) Angiography of a 53-year-old woman who pre-sented with severe claudication pain. Images at the iliac level show a type 2a PSA in combination with a small aneurysm. (b). Images at femoral level show a hypoplastic superficial femoral artery. Distally, the popliteal artery is occluded.

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and their summary. Manual cross-referencing was per-formed. All cases were analysed and summarised.

Results

From 1964 to 2007, 159 PSAs were described in 122 patients. Of these, 13 were found during post-mortem anatomical dissection in eight cases. These cases have been included only in the analysis of demographic data and classification. The ages ranged from a 32-week-old foetus e that was found to have PSA during post-mortem dissection e to 84 years, with a mean of 57 years. The PSA was diagnosed in 68 women (56%) and in 54 men (44%).

The PSA was unilateral in 70% of the patients (85 out of 122) and bilateral in 30% (37 out of 122). A unilateral PSA was equally present on the right and left side (43 and 41, respectively). In one unilateral PSA, the side was not mentioned. Using the classification provided by Bower et al.,1464 unilateral PSAs were classified as the complete type and 13 as the incomplete type. In the remaining eight cases, the type was not reported and could not be derived from the data. In the bilateral cases, the complete type was also the most common type. In 29 patients, the PSA was complete on both the sides, and, in three bilateral patients, the PSA was incomplete on both the sides. In three others, there was a difference between the right and the left side. In these cases, the PSA was of the complete type in one leg and of the incomplete type in the other. In two cases, the classification was not reported and could not be derived from the data.

Clinical features

Clinical data could be analysed from the 146 PSAs in 114 patients. The remaining eight cases were those from post-mortem dissections, excluded from further evaluation. Of the 114 cases, 82 presented with a unilateral PSA and 32 with a bilateral PSA, whereas 91 patients (80%) presented with symptoms of the PSA, including claudication, rest pain, necrosis, mass formation and neurological problems, and 23 patients (20%) were asymptomatic. These patients under-went evaluation for other medical problems (Table 1). In 19 out of 32 patients with a bilateral PSA, only one side was symptomatic at the time of presentation, while in four patients both sides were symptomatic. In eight patients both sides were asymptomatic, and in one case symptoms were not described.

Most patients went to the hospital because of arterial insufficiency and/or mass formation in one or both legs. The clinical presentation has been summarised inTable 2. Thirteen patients reported a combination of complaints. In the majority of patients (48 out of 91), the onset of symptoms was either sub-acute or chronic. In 24 out of 91 cases, the symptoms had an acute onset. In 19 others, the onset of symptoms was not reported.

Diagnostic tools

Cowie’s sign e described as a diminished or absent femoral pulse in combination with a palpable popliteal pulse e is considered to be pathognomic for a PSA58 and was

described in only five patients. In most cases, more than one diagnostic tool was used to evaluate the diagnosis. The diagnostic tests that were used have been summarised in

Table 3. Angiography (98 out of 114) was performed in most cases. In four cases, no diagnostic evaluation was per-formed because the condition of the extremity urged immediate surgical intervention.

Observations

In 35 out of 146 patients, a PSA was found without addi-tional pathology, such as stenosis or aneurysm formation. Sixteen of these were incidentally found during medical examination for other pathology and the others were found in patients with a contralateral symptomatic PSA. An aneurysm of the PSA was found in 48% (70 out of 146). In 27 of these, it was suspected that the aneurysm had caused distal embolisation. These embolisms were located in the PSA itself or in any other artery distally. In two additional

Table 1 Diagnoses in patients in whom an asymptomatic PSA was unexpectedly found

Medical problem Frequency Reference number

Varicoses 5 15e19

Gunshot 3 20e22

Hemi-hypertrophy leg 2 15,23

Postoperative bleeding 2 24,25

Abdominal pain (AAA) 2 26,27

Lung embolism 1 28

Chest pain 1 29

Rheumatoid arthritis 1 30

Mullerian agenesis 1 31

Fluxus 1 32

Evaluation haemodialysis access 1 33

Kidney transplant 1 34

Fracture femur after fall 1 35

Neurosarcoma left leg 1 36

Spina bifida, tethered cord and pelvic myelolipoma

1 13

Table 2 Symptoms at presentation of patients with symptomatic PSA Symptom Number of patients Reference number Arterial insufficiency Fontaine II 25 25,29,37e54

Fontaine III 27 9,24,33,39,51,55e73, case Fontaine IV 7 14,39,63,74e76

Mass per buttock

Painful 19 18,43,59,69,74,77e88

Painless 7 2,18,89e92

Pulsatile 8 51,62,64,74,82,93e95

Radicular pain 10 18,45,51,69,77,81,86,95e97

Footdrop 4 74,98

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cases, a pseudo-aneurysm was present in the PSA. In 7% (10 out of 146) of the PSAs, a stenosis was found; while in 9% (13 out of 146), an occlusion of the PSA was observed. In nine cases, there was an occlusion of an artery distal from the PSA. In one additional case, an occlusion was observed after a fracture of the femur. In six cases, hypoplasia of the SFA in combination with insufficient collateral filling was considered the cause of the symptoms.

Treatment

The choice of treatment mainly depended on symptoms and classifications of the PSA. Some patients received more than one treatment. In the 35 patients with a PSA without additional pathology, 26 patients did not receive any treatment. Irrespective of the absence of symptoms, a bypass was created in two patients, and in one patient the PSA was ligated. Treatment was not reported in five cases.

In the 10 patients with stenosis of the PSA, three were treated with angioplasty; a bare stent was placed in one of these cases; others were treated with a bypass (n Z 3), surgical thrombectomy (n Z 2) and chemical thrombolysis (n Z 2) and one patient was treated conservatively with anticoagulants. In one patient, the treatment was not described.

In four patients with an occlusion of the PSA, a bypass was created and in three, the PSA was ligated. The treat-ment was conservative with anticoagulants in three patients and an amputation was required in another patient. Acute surgical exploration was indicated in a patient because the condition of the leg deteriorated suddenly. During this exploration, a thrombectomy was performed and anticoagulants were started. The treatment was not documented in one patient. The treatment in the group with an occlusion in a distal vessel of the PSA was as follows: two patients received a bypass and chemical thrombolysis was also performed twice, one patient was treated conservatively with anticoagulants and another did not receive any treatment and in three cases the treatment was not described.

In 16 patients with an aneurysm of the PSA without distal occlusion, the aneurysm was ligated and a bypass was

created, whereas in 12 other patients only a ligation was performed and four patients received a bypass alone. In five cases, the aneurysm was occluded using coils and a stent-graft was placed in four patients. Surgical explo-ration followed by an amputation was reported once. In one other patient, an amputation was necessary. Aneurysmor-rhaphy and thrombolysis were both reported once. One patient was treated conservatively. In two cases, the treatment was not described.

The patients who were diagnosed with an aneurysm with a distal occlusion were treated by a ligation with a bypass in 15 cases. In one of these patients, amputation of the toes was necessary. Surgical thrombectomy was performed in six patients, while chemical thrombolysis was the treatment of choice in four patients. A stent-graft was placed in two patients and an amputation was required in five patients. Embolisation of the aneurysm was only required in one case as the treatment of choice. Two patients were treated conservatively and in one case the treatment was not described.

Two of the six patients with a hypoplasia of the SFA received a bypass. An amputation was required in one patient, and there was no treatment given in two patients. In one case, the treatment was not documented. A pseudo-aneurysm was bypassed in one case and another patient did not receive any treatment. The patient with the occlusion of the PSA after the fracture of the femur was treated for his fracture, but not for the occlusion.

Follow-up

Unfortunately, follow-up has been poorly described in most articles. After a follow-up of 6e12 months, 56 patients were reported to be in a good clinical condition and eight patients were relieved of their symptoms; nine patients had to adjust to life after an amputation of one extremity; in three patients, the footdrop was irreversible as a result of the pressure of the aneurysm on the sciatic nerve and one patient had chronic radicular pain. One asymptomatic patient, who was treated for carcinoma of the cervix 1 year earlier, died of massive bleeding from the PSA. Of the remaining 36 cases, the follow-up is not described in the articles and is, therefore, not known.

Discussion

In the present study, we have shown that a PSA is a rare anomaly with a high incidence of complications such as aneurysm formation and ischaemia. These complications may lead to an amputation in 8% of the cases. It seems reasonable to assume that most asymptomatic PSAs are not discovered. Based on the described cases, a strategy for follow-up cannot be extracted from the literature.

A large variety of diagnostic tests may confirm the diagnosis of a PSA. Angiography is the most frequently used diagnostic tool to detect a PSA. It provides information about the classification and outflow of the PSA, which is important with regards to treatment. A computed tomo-graphic (CT) or magnetic resonance imaging (MRI) scan may show the relation of the PSA e and possible aneurysm e with other structures such as the sciatic nerve. This is

Table 3 Diagnostic tests used for the evaluation of a PSA Diagnostic tool Frequency Reference number

Angiography 115 2,9,13e25,27e29,31e38, 40e44,46,52,54e76,78e92, 94,95,97, case CT/CTA 30 25,27,30,36,39,40,46,49,56, 57,60,62,65,67,77,78,82, 88,90,94,97 MRI/MRA 12 9,24,49,56,77,81,83,90,96,98 Duplex 9 15,16,37,38,40,77,79,89, case Doppler 4 21,38,43,50 Ultrasound 7 24,78,83,85,96,98 Phlebography 2 15,16 None 1 51 Not reported 4 53,74

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important because the close relation of the PSA with the sciatic nerve has surgical consequences. In a small retro-spective study in five patients with a PSA, it was shown that CT angiography might be used as the only imaging tech-nique for a PSA.39 CT angiography may also demonstrate a totally occluded PSA that cannot be visualised with conventional angiography. Other diagnostic tools such as duplex scanning may be helpful, but may not provide sufficient information.

Aneurysm formation is the most frequent complication of a PSA, occurring in 48% of all PSAs. Generally, the aneurysm develops in the buttocks between the pyriform muscle and the posterior aspect of the greater trochanter of the femur7ethe site where the PSA has a slight S-shaped curve.7 Aneurysms in a PSA may lead to severe

complica-tions, including thrombosis, embolisation and radicular pain. The latter may be caused by compression of the sciatic nerve,23,29,78 which has a close relation with the PSA. The exact reason why an aneurysm is frequently seen in a PSA is unknown. Possible causes may be overstretching, as a consequence of the anatomical position of the PSA. Another hypothesis is that the PSA is pressed against the sacrospinal ligament, pyriform muscle and hip during flexion of the hip joint.59Other studies have suggested that

a reduced concentration of elastic material in a congeni-tally impaired arterial wall,79or atherosclerosis,93may also

play a role. In early cases, an association with syphilis has been suggested.99 Stenosis and occlusions of the PSA are also frequently reported, with an incidence of 7% and 9%, respectively.

Treatment of a PSA is mainly dependent on the symp-toms of the patients. Surprisingly, we found that three patients with an asymptomatic PSA were surgically treated. In our opinion, surgical intervention is not indicated in the absence of symptoms. Although evidence-based follow-up strategies are lacking, regular control with physical exam-ination, ankleebrachial index (ABI) and duplex scanning seem appropriate. In case of an aneurysm, the PSA may be ligated when the SFA provides sufficient blood to the distal extremity.44,69,92This may be an option when the PSA is of

the incomplete type. When the SFA is hypoplastic, however, an additional bypass is needed to maintain the blood flow to the lower extremity. When excision of the sciatic aneurysm is combined with an interposition graft, the patency may be compromised when the patient sits down.29 Exclusion of a sciatic aneurysm may also be complicated by sciatic nerve damage. Endovascular treat-ment, including coiling or the placement of a stent-graft, may be a good alternative when the PSA is stretched over the sciatic nerve. Another alternative is to leave the aneurysm wall intact during exclusion.61

In some cases, the PSA is found in combination with other anomalies such as Mullerian agnesis,31arteriovenous fistulae,100hypertrophy of the lower limb19,23and varicose

veins in the young with an atypical distribution.17,19,23,100 The PSA may also exist in combination with a suprafacial brachial artery101and a right retro-oesophageal subclavian artery.102 A single genetic or environmental event in the

early vascular development has been suggested as the cause of combined vascular anomalies.19The co-existence with a hypertrophic limb may be explained by the fact that both the vascular and the musculoskeletal system are

derived from the mesoderm. A defect in the mesoderm may affect both systems.19

In conclusion, the PSA is a rare vascular anomaly with a high incidence of complications such as aneurysm formation and ischaemia that may lead to amputation. Treatment options depend on the classification and symp-tomatology of the individual patient. Strategies for follow-up in asymptomatic PSAs cannot be deduced from the literature.

Conflict of interests/funding

None.

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