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Arterial endofibrosis in professional cyclists

  • University Hospital of Verona


External Iliac Artery Endofibrosis (EIAE) is an uncommon disease usually affecting young, otherwise healthy, patients. It usually involves cyclists but cases have been reported in other groups of endurance athletes. The external iliac artery is the most affected anatomical site but other locations are described too. The precise pathophysiology and long-term evolution of the disease still remain unknown. The diagnosis may be challenging and delayed as the patients usually present symptoms only in extreme conditions and physical and instrumental examinations may be normal at rest. We present two cases of young professional cyclists who suffered of exercise-induced leg pain which led them to reduce running. Both patients were firstly treated with balloon angioplasty that rapidly failed to improve their symptoms. The successive open surgery with endofibrosectomy and autologous saphenous vein closure patch completely resolved physical limitations. EIAE is a rare disease that can induce arterial stenosis, thrombosis, dissection and secondary atheroma. After-exercise ankle-brachial index represents a useful diagnostic criterion. Careful observation of angio-CT may strengthen the suspect. Knowledge of the these features allows a better pre-operative assessment and an early effective treatment. Surgical revascularization remains the gold standard approach.
External Iliac Artery Endofibrosis (EIAE) is an un-
common and rare disease usually affecting primarily
young, otherwise healthy, athletes. The first description
of the disease was by Chevalier in 1986 in cyclists (1)
but cases have been reported in other groups of endu-
rance athletes including triathletes, runners, cross-
country skiers, rowers and rugby players (2).
The external iliac artery is the most affected site but
other locations have been reported (3-5). The precise
pathophysiology and long-term evolution are unk-
nown. The diagnosis may be difficult and often delayed
due to the absence of signs and symptoms at rest.
We present two cases of young professional cyclists
who suffered of exercise-induced leg pain significantly
reducing their physical performance and we discuss on
this entity whose knowledge is crucial for correct diagnosis
and treatment.
Case 1
A 23-year-old professional cyclist was referred for mu-
scle fatigue with significant cramping and pain of his ri-
ght leg when attempting to run. He was in optimal ge-
neral conditions with a complete negative medical hi-
Clinical examination revealed that common femo-
ral pulses were both palpable and strong; otherwise the
distal pulses were clearly palpable only in the left leg. The
skin was bilaterally warm without any pathological sign.
The ankle-brachial index (ABI) at rest and after exer-
cise was performed. Resting-ABI was 1.02 on the right
side and 1.04 on the left. After exercise right ABI drop
to 0.61 while left ABI remained the same.
SUMMARY: Arterial endofibrosis in professional cyclists.
External Iliac Artery Endofibrosis (EIAE) is an uncommon disea-
se usually affecting young, otherwise healthy, patients. It usually invol-
ves cyclists but cases have been reported in other groups of endurance
athletes. The external iliac artery is the most affected anatomical site
but other locations are described too. The precise pathophysiology and
long-term evolution of the disease still remain unknown. The diagno-
sis may be challenging and delayed as the patients usually present symp-
toms only in extreme conditions and physical and instrumental exami-
nations may be normal at rest.
We present two cases of young professional cyclists who suffered of
exercise-induced leg pain which led them to reduce running. Both pa-
tients were firstly treated with balloon angioplasty that rapidly failed to
improve their symptoms. The successive open surgery with endofibro-
sectomy and autologous saphenous vein closure patch completely resol-
ved physical limitations.
EIAE is a rare disease that can induce arterial stenosis, thrombo-
sis, dissection and secondary atheroma. After-exercise ankle-brachial
index represents a useful diagnostic criterion. Careful observation of
angio-CT may strengthen the suspect. Knowledge of the these features
allows a better pre-operative assessment and an early effective treat-
ment. Surgical revascularization remains the gold standard approach.
KEY WORDS: External iliac artery endofibrosis - Peripheral arterial disease - Non atherosclerotic arterial disease.
Arterial endofibrosis in professional cyclists
G Chir Vol. 36 - n. 6 - pp. 267-271
November-December 2015
Department of Vascular Surgery, Polo Chirurgico “Pietro Confortini”,
University Hospital of Verona, Verona, Italy
Corresponding author: Gian Franco Veraldi,
© Copyright 2015, CIC Edizioni Internazionali, Roma
clinical practice
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 267
G.F. Veraldi et al.
The subsequent angio-CT documented a normal
common iliac artery on the right; on the contrary, a mi-
nimal calibre reduction of the proximal tract of right ex-
ternal iliac artery (rEIA) was identified with no other si-
gnificant radiological lesions (Figure 1).
An endovascular approach was attempted firstly: by
means of a retrograde femoral access, an invasive pressure
examination in right common iliac artery under and abo-
ve the proximal segment of external iliac artery was ob-
tained, without significant difference, even after papaverine
intra-arterial injection. After the pressure study, we perfor-
med an angioplasty of the EIA with a 10-millimeter dia-
meter non-compliant balloon for at least 3 minutes of
inflation. Post-procedural period was regular.
After discharge, the patient did not refer any clinical
improvement and an open surgical repair was planned.
The right iliac vessels were exposed by mean of a
right pararectal extra-peritoneal approach. Longitu-
dinal arteriotomy of rEIA at site of lumen reduction
revealed a significant endothelial hyperplasia. An en-
dofibrosectomy (Figure 2) was performed and the ar-
tery was closed with an autologous great saphenous
vein patch.
Histological analysis revealed marked fibrosis of the
tunica media. The subsequent hospital stay was une-
ventful. The patient was discharged at fourth post-ope-
rative day.
Two months later the patient returned to ride the bike
with no more symptoms and after 6 months he won a
professional cyclist race.
One-year angio-CT follow up confirmed the regu-
lar patency of the rEIA lumen.
Fig. 1 A, B - Case 1: CT-angiography showing a minimal calibre reduction of the right external iliac artery compared to the left external iliac artery with no
other evident lesions (A: 3D-lumen reconstruction, B: MIP-reconstruction with EIA diameter indicated).
Fig. 2 A and B - Case 1: endofibrosectomy of the origin of the right external iliac artery.
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 268
Case 2
A 25-year-old professional cyclist presented a clini-
cal history very similar to the case one.
The CT-scan revealed a narrowing at the origin of
the left external iliac artery (lEIA), in contact to the left
psoas muscle that appeared hypertrophic.
At another Institution, a balloon angioplasty was fir-
stly attempted, followed by laparoscopic debridment of the
lEIA from the left psoas muscle. After these two attempts,
he presented at our Vascular Institution referring an im-
mediate recurrence of disabling symptoms. The clinical exa-
mination at rest did not reveal any alteration with normal
peripheral pulses. Anyway ABI examination before and
after exercise revealed a significant drop on left side (from
1 to 0.60). In consideration of previous failed mininvasi-
ve approach, we managed for open revascularization.
The left iliac vessels were exposed by means of a left
pararectal extra-peritoneal approach. Tough adhesions
between the artery and the hypertrophic psoas muscle
were identified, narrowing the origin of the external iliac
artery (Figure 3 A). After lysis of the adhesions and a com-
plete dissection of the iliac vessels from neighbourough
tissue (Figure 3 B), an endofibrosectomy of the first tract
of the lEIA and an autologous great saphenous vein clo-
sure patch was performed (Figure 4).
Even in this case, the histological examination revealed
a severe fibrosis of the tunica media.
Arterial endofibrosis in professional cyclists
Fig. 3 A, B - A) Case 2: it is evident the narrowing of the origin of the external iliac artery due to the severe ipertrophy of the left psoas muscle. B) The left
iliac vessels after complete separation from the psoas muscle.
Fig. 4 - Case 2: autologous saphenous vein clo-
sure patch.
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 269
The postoperative course was uneventful and the pa-
tient was discharged at fifth post-operative day.
After the treatment, the patient’s symptoms imme-
diately disappeared and after two months he returned
to ride a bike at professional levels.
Eight-month angio-CT showed restoration of the
lEIA lumen with no vascular complications.
External Iliac Artery Endofibrosis (EIAE), although un-
common, represents an important clinical entity affecting
cyclists and other endurance athletes otherwise healthy.
The prevalence of the disease is unknown but in profes-
sional cyclists it can be estimated to account up to 20%
of all overuse leg injuries (3). EIAE has also been repor-
ted in other endurance sports such as long-runners, triath-
letes, speed skating and more (4-6). Although the exter-
nal iliac artery is mainly affected, the common iliac and
the common femoral arteries may also be involved (3-5).
Certainly different from the atherosclerosis, the ae-
tiology of EIAE still remains unclear. In histological spe-
cimens there is a thickening of the vessel intima due to
subendothelial accumulation of loose connective tissue
containing variable amounts of collagen, elastin and
smooth muscle cells, as shown also in our cases. Diffe-
rently to the atherosclerosis mechanism, the collagen fi-
bres are densely packed and calcification is typically wi-
despread (7-9).
The pathophysiology has been investigated in the cy-
clist patients and some factors have been postulated. Fir-
st of all, the position: in order to obtain the maximal ae-
rodynamic benefit, they force an hyperflexion of the hip
joint that may alter the iliac artery anatomy inducing a
chronic friction and stimulating the endofibrosis (9). A
significant hypertrophy of the psoas muscle may wor-
sen the mechanical stress: the muscle pinches the artery,
which is often fixated to it by the fascia and its collate-
ral branches, provoking a continuous arterial traction and
a subsequent endofibrosis (9, 10). This mechanism was
particularly evident in our second case, where a thigh
adhesion between artery and muscle was found.
The diagnosis may be difficult and delayed. Usual-
ly, symptoms are unilateral and they appear only at near-
maximal exercise. In that situation, muscle cramp is the
most frequent symptom followed by feeling of swelling,
numbness or pain that develops in the calf, thigh or but-
tock of the affected side (5, 11, 12). At rest, physical exa-
mination is generally completely negative and periphe-
ral pulses are present with absence of the traditional si-
gns of arterial disease. Different dynamic instrumental
investigations (i.e. ABI measurement, pulse volume re-
cording and papaverine-assisted mean pressure gradient)
have been proposed to help in the diagnosis. In our ex-
perience, the ABI measurement at rest and after exerci-
se resulted positive (at least 0.4 of difference) and it cla-
rified many diagnostic doubts. It probably represents one
of the easier, cheaper and more effective methods to in-
vestigate this rare arterial disease, especially when asso-
ciated to uncertain clinical or radiological finds (6-8).
The predictive value of Duplex, in fact, remains low even
in trained hands and a normal Duplex should not ex-
clude the diagnosis. Angio-CT well reveals stenosis, dis-
section or arterial lumen narrowing but it requires ca-
reful attention because of arterial defects may be mini-
mal, as in our first case. The role of MR-angiography is
limited to those patients without endovascular lesions
whose symptoms may be due to arterial kinking. Digi-
tal subtraction angiography provides anatomic details
before surgical intervention and allows location of the
stenosis and quantification of the excessive length of the
artery (2, 13).
Surgery is the best treatment in individuals who want
to continue their sporting lifestyle. The most common
method of repair is endofibrosectomy with patch an-
gioplasty (1). In more complex cases reconstruction with
either autologous or prosthetic interposition graft has been
documented to have excellent results, with 90% primary
patency and 99% return to sport, including return to
high-level competition (2).
Transluminal balloon angioplasty and stenting te-
chniques have both been proposed but they are not con-
sidered appropriate for treating endofibrotic lesions. Un-
like atherosclerotic stenosis, these lesions are elastic and
tend to re-coil or dissect within days from the procedure
(4). Moreover, a stent placed in the external iliac artery
would be subject to the same forces and deterioration
of the native artery if the patient return to the same le-
vel of activity. Potential fracture, plicature or migration
of the stent were all concerns. Long-term outcome of stent
angioplasty in such condition remains debatable (9).
EIAE is a rare disease which could give to the clini-
cians both diagnostic and therapeutic problems. Clini-
cal history, physical examination in conjunction with pre-
and post-exercise ABI and careful observation of angio-
CT represent the fundamental criteria to establish the
correct diagnosis. Once diagnosed, surgical endofibro-
sectomy with arterial patch closure is the best treatment
for obtaining a definitive clinical benefit.
Conflict of interest
The Authors declare that there is not conflict of interest.
G.F. Veraldi et al.
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 270
Arterial endofibrosis in professional cyclists
1. Chevalier JM, Enon B, Walder J, Barral X, Pillet J, Megret A,
et al. Endofibrosis of the external iliac artery in bycicle racers:
an unrecognized pathological state. Ann Vasc Surg. 1986;1:297-
2. Wilson TD, Revesz E, Podbielski FJ, Blecha MJ. External iliac
artery dissection secondary to endofibrosis in a cyclist. J Vasc Surg.
3. Schep G, Schmikli SL, Bender MH, Mosterd WL, Hammacher
ER, Wijn PF. Recognising vascular causes of leg complaints in
endurance athletes. Part 1: validation of a decision algorithm.
Int J Sports Med. 2002;23:313-321.
4. Bender MH, Schep G, Bouts SW, Backx FJ, Moll FL. Endurance
athletes with intermittent claudication caused by iliac artery ste-
nosis treated by endarterectomy with vein patch—short- and mid-
term results. Eur J Vasc Endovasc Surg. 2012;43:472-477.
5. Ford SJ, Rehman A, Bradbury AW. External iliac endofibrosis
in endurance athletes: a novel case in an endurance runner and
a review of the literature. Eur J Vasc Endovasc Surg. 2003;26:629-
6. Maree AO, Ashequl Islam M, Snuderl M, Lamuraglia GM, Sto-
ne JR, Olmsted K, et al. External iliac artery endofibrosis in an
amateur runner: hemodynamic, angiographic, histopathologi-
cal evaluation and percutaneous revascularization. Vasc Med.
7. Rousselet MC, Saint-Andre JP, L’Hoste P, Enon B, Megret A, Che-
valier JM. Stenotic intimal thickening of the external iliac artery
in competition cyclists. Hum Pathol. 1990;21(5):524e9.
8. Vink A, Bender M, Schep G, van Wichen D, de Weger R, Pa-
sterkamp G, et al. Histopathological comparison between en-
dofibrosis of the high-performance cyclist and atherosclerosis in
the external iliac artery. J Vasc Surg. 2008;48(6):1458-63.
9. Peach G, Schep G, Palfreeman R, Beard JD, Thompson MM,
Hinchliffe RJ. Endofibrosis and kinking of the iliac arteries in
athletes: a systematic review. Eur J Vasc Endovasc Surg.
10. Lindner D, Agar G, Domb BG, Beer Y, Shub I, Mann G. An
unusual case of leg pain in a competitive cyclist: a case report and
review of the literature. Sports Health. 2014;6(6):492-496.
11. Del Gallo G, Plissonnier D, Planet M, Peillon C, Testart J, Wa-
telet J. Dissecting aneurysm of the external iliac artery. An unu-
sual course of endofibrosis in an athlete [in French]. J Mal Vasc.
12. Kral CA, Han DC, Edwards WD, Spittell PC, Tazelaar HD,
Cherry KJ Jr. Obstructive external iliac arteriopathy in avid bicy-
clists: new and variable histopathologic features in four women.
J Vasc Surg. 2002;36:565-570.
13. Flors L, Salinas-Leiva C, Bozlar U, Norton PT, Cherry KJ, Hous-
seini AM, et al. Imaging evaluation of flow limitations in the iliac
arteries in endurance athletes: diagnosis and treatment follow-
up. AJR. 2011;197:W948-W955.
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 271
... Arterial endofibrosis is a vascular disease afflicting primarily the external iliac artery. It presents mostly in cyclists as leg weakness and thigh pain on exertion [12]. Unlike PAD which constitutes the presence of atherosclerotic plaque, arterial endofibrosis is characterized by the buildup of loose connective tissue within the tunica intima of arterial walls [12]. ...
... It presents mostly in cyclists as leg weakness and thigh pain on exertion [12]. Unlike PAD which constitutes the presence of atherosclerotic plaque, arterial endofibrosis is characterized by the buildup of loose connective tissue within the tunica intima of arterial walls [12]. The etiology or inciting events behind arterial endofibrosis still remains unknown. ...
... However, digital subtraction angiography allows the identification of the location of the stenotic area prior to surgical intervention. CT angiography may reveal the stenosis; however, some patients have been noted to have minimal arterial endofibrosis which was not identified on CT angiography [12]. ...
Popliteal artery entrapment syndrome (PAES) is a type of arterial obstruction seen in athletic and young patients with no cardiovascular risk factors. It is caused by aberrant anatomy affecting the position of the popliteal artery or gastrocnemius muscle or functional obstruction resulting from a hypertrophied gastrocnemius muscle. Rich's classification has been used to define the various entities. PAES presents as unilateral claudication exacerbated by physical exertion. However, such a clinical presentation is shared amongst not only vascular diseases but also musculoskeletal diseases. Therefore, a wide array of differential diagnoses must be considered when popliteal artery entrapment-induced claudication is suspected.
... Arterial endofibrosis is a non-atherosclerotic cause of exertional leg pain in young athletes, particularly cyclists [25]. It primarily afflicts the external iliac artery [26]. The exact pathophysiology of the disease is unknown but there is suspected to be an association between duration of time spent cycling and the development of ▶Fig. 1 Subtypes of Popliteal Artery Entrapment Syndrome (PAES). ...
... the disease [25,26]. Typical symptoms include leg weakness and thigh pain on exertion, which resolve with cessation of exercise. ...
Popliteal Artery Entrapment Syndrome (PAES) is an uncommon syndrome that predominantly affects young athletes. Functional PAES is a subtype of PAES without anatomic entrapment of the popliteal artery. Patients with functional PAES tend to be younger and more active than typical PAES patients. A number of differential diagnoses exist, the most common of which is chronic exertional compartment syndrome. There is no consensus regarding choice of investigation for these patients. However, exercise ankle-brachial indices and magnetic resonance imaging are less invasive alternatives to digital subtraction angiography. Patients with typical symptoms that are severe and repetitive should be considered for intervention. Surgical intervention consists of release of the popliteal artery, either via a posterior or medial approach. The Turnipseed procedure involves a medial approach with a concomitant release of the medial gastrocnemius and soleal fascia, the medial tibial attachments of the soleus and excision of the proximal third of the plantaris muscle. Injection of botulinum A toxin under electromyographic guidance has recently shown promise as a diagnostic and/or therapeutic intervention in small case series. This review provides relevant information for the clinician investigating and managing patients with functional PAES.
... For example, it is possible to investigate regulatory mechanisms or organs interplay as the relationship between acute kidney injury and some adverse cardiac dysfunctions (66). Another example comes from professional cycling: arterial endofibrosis is a common pathology in cyclists (67) and it led to clinical improvement in its diagnosis and treatment (68). (71); warm bath (72) for heat acclimatization); full body cryotherapy for improving recovery or rehabilitation (73)…]. ...
Full-text available
Lowering of the upper body to optimize cycling time trial (TT) performance is a balance between the aerodynamic advantage related to a lower frontal area and prospective detrimental physiological effects associated with a reduction of the hip-torso angle. To explore this in elite athletes and across positions relevant for competitive cyclists, we analysed racing positions for world championships [WC] top-10 finishers and 10 national elite TT-cyclists. Subsequently, laboratory studies were completed to evaluate effects on exercise economy, muscle oxygenation and perceived exertion for the national TT-group for their habitual position and compared to standard (4-12-20˚) torso angles. Hence, covering the racing position observed for top-10 WC finishers (positioned from 4-12˚) and the national elite (range 8-18˚). Oxygen calorimetry and near-infrared spectroscopy revealed that there was no difference in overall energy expenditure, delta exercise efficiency or muscle oxygenation across the investigated range of positions. However, rating of perceived exertion was significantly elevated for the lowest position (4˚ torso angle) compared to the rider’s habitual position. This lets us conclude that elite TT-cyclists can acutely adopt to a very low upper body position without compromising exercise economy or muscle oxygenation and some WC-level TT riders have adopted this low (4˚) racing position. However, the elevated perception of exertion with an acute reduction of the torso-hip angle indicates that it presumably requires specific training in the position or factors not related to exercise economy and muscle oxygenation determine if a rider in practice can perform in the very low position.
Introduction External iliac artery endofibrosis (EIAE) classically presents in cyclists with intimal thickening of the affected arteries. We investigated possible anatomical predisposing factors including psoas muscle hypertrophy, arterial tortuosity, inguinal ligament compression, and arterial kinking via case-control comparison of symptomatic and contralateral limbs. Methods All patients with unilateral EIAE treated surgically at our institution were reviewed. Each patient’s symptomatic side was compared with their contralateral side using paired t-tests. Psoas hypertrophy was quantified by transverse cross-sectional area (CSA) at L4, L5, and S1 vertebral levels, and inguinal ligament compression was measured as anterior-posterior distance between inguinal ligament and underlying bone. Tortuosity index for diseased segments and arterial kinking were measured on TeraRecon. Results Of 33 patients operated on for EIAE from 2004-2021, 27 with available imaging presented with unilateral disease, more commonly left-sided (63%). Most (96%) had external iliac involvement and 26% had ≥2 segments affected: 19% common iliac artery, 15% common femoral artery. The symptomatic limb had greater mean L5 psoas CSA (1450 mm² vs. 1396 mm², mean difference 54 mm², P=0.039). There were no significant differences in L4 or S1 psoas hypertrophy, tortuosity, inguinal ligament compression, or arterial kinking. 63% underwent patch angioplasty and 85% underwent additional inguinal ligament release. 84% reported postoperative satisfaction, which was associated with greater difference in psoas hypertrophy at L4 (p=0.022). Conclusions Psoas muscle hypertrophy is most pronounced at L5 and is associated with symptomatic EIAE. Preferential hypertrophy of the affected side correlates with improved outcomes, suggesting psoas muscle hypertrophy as a marker of disease severity.
We present a case of a 30-year-old female professional cyclist who presented with right thigh claudication while performing high-intensity efforts. Based on the typical clinical history and the presence of a smooth long segment diffuse narrowing in the right external iliac artery on non-contrast magnetic resonance angiography, a diagnosis of external iliac artery endofibrosis or "cyclist's iliac syndrome" was made. Supplementary information: The online version contains supplementary material available at 10.1007/s12055-022-01349-8.
External iliac vein stenosis related to cycling has rarely been reported as a cause of deep vein thrombosis. Ischemic stroke occurring in this condition due to paradoxical embolism across a preexisting patent foramen ovale (PFO) has yet to be reported. Here we report a case of embolic ischemic strokes in a young, avid cyclist with no prior known vascular risk factors. A thorough cerebrovascular workup revealed a right-to-left shunt on transesophageal echocardiogram that prompted venous thrombosis evaluation. Pelvic MR venogram demonstrated a 3.5 cm high-grade stenosis of the right external iliac vein, concerning for possible prior thrombotic disease. His strokes were deemed most likely a result of paradoxical emboli originating in the pelvis at the site of right external iliac vein stenosis. The patient ultimately opted for PFO closure for secondary stroke prevention, as he wished to continue daily cycling. This case highlights the importance of neurohospitalists considering iliac vein stenosis as a potential cause of embolic stroke of undetermined source, especially in young patients who are avid cyclists, as part of a thorough vascular workup.
Popliteal compression syndromes are a rare cause of vascular pathology and should be considered in the differential diagnosis of young athletic men with claudication symptoms. The underlying cause is compression by hypertrophied muscles (functional entrapment) or aberrant courses of the vessels or surrounding muscle or tendon structures. The median time from onset of symptoms to diagnosis is 12 months. Permanent damage to the arterial wall with stenosis or occlusions up to poststenotic aneurysm formation with thromboembolic occlusions can be the consequence. Diagnostically, popliteal artery entrapment syndrome (PAES) is challenging and often unrecognized. While clinical symptoms with intermittent claudication, calf/foot pain or dysesthesia are usually non-specific, functional tests with loss of pulse during dorsiflexion/plantar flexion or a weakened ABI after exercise can be indicative. Functional tests, such as duplex sonography and magnetic resonance angiography (MRA) are useful and digital subtraction angiography is useful in the case of inconclusive findings. Surgical treatment is necessary in symptomatic PAES. The prognosis is favorable with a timely diagnosis but vascular reconstruction is required if damage to the vessel wall has already occurred.
Full-text available
Infection of prosthetic vascular grafts can manifest as pain, pseudoaneurysms, or arterial insufficiency in the leg. We present the case of a female patient with a medical history of a right external iliac artery endofibrosis, with a persistently infected synthetic iliofemoral bypass graft, replaced with a bioengineered human acellular vessel. At the 12-month post-implantation visit, clinical and radiological studies demonstrated adequate human acellular vessel patency, with no signs of infection, stenosis, or pseudoaneurysm. Subsequent to the initiation of hormone therapy and cessation of anti-platelet therapy, the patient suffered graft thrombosis. She continues to do well after restoration of patency with lytic therapy. At 22 months, secondary patency is maintained with continued anticoagulation and the patient remains asymptomatic.
Cycling has become a popular recreational and competitive sport. The number of people participating in the sport is gradually increasing. Despite being a noncontact, low-impact sport, as many as 85% of athletes engaged in the sport will suffer from an overuse injury, with the lower limbs comprising the majority of these injuries. Up to 20% of all lower extremity overuse injuries in competitive cyclists are of a vascular source. A 39-year-old competitive cyclist had a 5-year history of thigh pain during cycling, preventing him from competing. The patient was eventually diagnosed with external iliac artery endofibrosis. After conservative treatment failed, the patient underwent corrective vascular surgery with complete resolution of his symptoms and return to competitive cycling by 1 year. Since its first description in 1985, there have been more than 60 articles addressing external iliac artery endofibrosis pathology.
Endurance athletes may suffer from intermittent claudication. A subgroup of 16% has severe iliac artery stenosis due to endofibrosis. In this study we report the short- and mid-term results of endarterectomy with venous patching. Athletes with claudication-like complaints were analysed using a protocol including cycling test and provocative echo-Doppler. Thirty-six athletes were diagnosed with serious iliac flow limitation (one bilateral), confirmed by additional magnetic resonance (MR) angiography. Endarterectomy with venous patching was performed for 32 iliac artery stenosis and five occlusions. Postoperative (mean 15.6 months) 33 legs were evaluated using the same diagnostic protocol. A complete follow-up after mean 29 months was obtained by questionnaire. Twenty-eight athletes were symptom free or could perform on a desired level with minor remaining complaints. Two athletes were satisfied though minor complaints prohibited high competition performance. Two athletes developed a re-stenosis and became symptom free after an additional operation. Three athletes had objective improvement but limited decrease in symptoms. One was unsatisfied but refused postoperative tests. The only major surgical complication was a postoperative bleeding necessitating re-operation. Postoperative tests showed significant increase in maximal workload and post-exercise ankle-brachial index. No aneurysm formation was detected. Precise diagnosis and meticulously performed endarterectomy with vein patching have satisfactory results in mid-term follow-up with acceptable risk in endurance athletes complaining of intermittent claudication due to iliac artery stenosis.
Kinking and endofibrosis of the iliac arteries are uncommon and poorly recognized conditions affecting young endurance athletes. Deformation or progressive stenosis of the iliac artery may reduce blood flow to the lower limb and adversely affect performance. The aim of this review was to examine the existing literature relating to these flow-limiting phenomena and identify a clear, unifying strategy for the assessment and management of affected patients. A systematic review of the literature was performed. A comprehensive search was carried out using Medline, Embase and The Cochrane Database to identify relevant articles published between 1950 and 2011 (last search date 05/08/2011). This search (and additional bibliography review) identified 413 articles, of which 367 were excluded. 46 articles were then studied in detail. Methodological quality of studies was assessed according to Scottish Intercollegiate Guideline Network criteria. Focussed history and examination can successfully identify nearly 80% of patients with iliac flow limitation. However, both provocative exercise tests and detailed imaging are also necessary to identify those in need of intervention and establish most appropriate treatment. Provocative exercise tests and duplex imaging can then be used to confirm flow limitation before detailed assessment of abnormal anatomy with MRA and DSA. These multiple imaging modalities are necessary to identify those most likely to benefit from surgery and clarify whether each patient should undergo arterial release, vessel shortening, endofibrosectomy or interposition grafting. We present a systematic review of the literature together with a proposed algorithm for diagnosis and management of these iliac flow limitations in endurance athletes.
Objective: The purpose of this article is to review the role of imaging in the diagnosis, treatment, and follow-up of patients with sport-related flow limitations in the iliac arteries. Conclusion: Endurance athletes can develop flow restriction during exercise because of endofibrosis or kinking of the iliac arteries. Knowledge of this entity and the use of appropriate imaging techniques are crucial for diagnosis. Imaging plays an important role in the assessment of the underlying lesion and its location as well as in posttreatment follow-up.
Endofibrosis of the external iliac artery is an uncommon disease affecting primarily young, otherwise healthy, endurance athletes. Thigh pain during maximal exercise with quick resolution postexercise is characteristic of the so-called cyclist's iliac syndrome. We report an unusual case in which the typical endofibrotic plaque was accompanied by dissection of the external iliac artery. The patient was treated surgically with excision of the affected artery segment and placement of an interposition graft. This case highlights an unusual finding in association with external iliac artery endofibrosis and provides an opportunity to briefly review the literature on the subject.
High performance athletes, predominantly professional cyclists, can develop symptomatic arterial flow restriction in one or both legs during exercise. The ischemic symptoms are caused by endofibrosis and/or kinking of the external iliac artery. Because these athletes are young and have no classic risk factors for atherosclerosis, endofibrosis and atherosclerosis are considered different disease entities. We compared histology of endofibrotic lesions from young sportsmen with atherosclerotic lesions of the external iliac artery in elderly individuals. Nineteen external iliac endarterectomy specimens from 18 cyclists (age 29 +/- 8 years) were compared with 42 external iliac segments from 22 elderly individuals (82 +/- 10 years). Ten arteries from elderly individuals revealed an intimal area that was >or=25% of the area encompassed by the internal elastic lamina and were considered atherosclerotic lesions. Stenosis was higher in patients (65% [interquartile range 50-75]) than in controls (11% [7-24]) (P < .0001). The endofibrotic lesions revealed loose connective tissue with moderate to high cellularity. Both in endofibrosis and atherosclerosis, most cells in the lesion were smooth muscle actin positive. In the endofibrosis specimens, loose fibers of collagen were observed, whereas in the atherosclerotic lesions collagen was mostly densely packed. Calcification of the lesion was not observed in endofibrotic lesions, whereas calcium deposition was observed in 80% of atherosclerotic lesions. Lymphocytes were present in 21% of endofibrotic lesions and in 80% of atherosclerotic cases. Macrophages were observed in 16% of endofibrotic lesions and in all atherosclerotic plaques. Luminal thrombosis was observed in one case of endofibrosis. In the external iliac artery, atherosclerotic lesions and endofibrotic lesions of high performance cyclists have distinct morphologic characteristics. Endofibrosis in the external iliac artery may serve as soil for luminal thrombosis.
Twenty-three cases of an arterial disease that affects competition cyclists are reported. Patients complained of intermittent acute claudication appearing on one lower limb only at the time of a maximal strain while cycling. Doppler hemodynamic investigation on an ergometric bicycle revealed a collapse of the ankle systolic pressure. Arteriography showed a sinuous lengthening and moderate stenosis of the external iliac artery. Pathologic examination of the artery disclosed a stenotic intimal thickening due to moderately cellular loose connective tissue with a variable distribution of collagen and elastic fibers. The cells in the affected zone were readily labeled with anti-actin and anti-myosin antibodies, and electron microscopy revealed features of synthetic smooth muscle cells. The lesion observed differs from intimal fibrodysplasia and from artherosclerosis. Abnormal local hemodynamic conditions may lead to this type of lesion. Thus, stenotic intimal thickening of the external iliac artery appears to be a new arterial disease defined by clinical, arteriographic, and pathologic features.
Since December 1985, we have operated upon seven bicycle racers for endofibrosis of the external iliac artery. In all instances, the athletes had started cycling early in life and were engaged in top level competition by the age of 17. The principal complaint was intermittent claudication of one lower limb at "near-maximal" exercise. Pain could be reproduced by exertional tests on an ergometric bicycle, and in all cases except one, measurement of ankle systolic pressure of that limb compared with the opposite side and brachial pressures showed a marked decrease on the involved side. Arteriography, performed with multiple views and positions, documented a 5 to 6 cm moderately stenotic (less than 40% diameter) segment, associated with arterial lengthening. Surgical treatment consisted of endarterectomy and shortening of the artery. Four patients were able to return to competition. The origin of this pathology is discussed, based on gross and histologic findings. Under certain predisposing anatomic conditions, abnormal hemodynamics, probably due to a high flow arterial state and an aerodynamic position on the bicycle, provoke repeated trauma which eventually produces the lesion. Practically unrecognized until now with only two previous publications on the subject in the literature, this entity is probably not uncommon.
A 42-year-old man was consulted because of a pain in his left leg. He was a highly trained biker since 20 years. The echo-Doppler and arteriography evidenced a stenosis, probably due to endofibrosis of the external iliac artery. In addition, it showed an aneurysm and an intimal dissection of this artery. The arteriography confirmed this diagnosis, and normal aspect of the other arteries. Neither conservative nor endovascular treatments were possible because of the anatomic lesions. We resected the external iliac artery and performed a by-pass with the great saphenous. The result at the 5th month was clinically good. The echo-Doppler control did not show any abnormality. The natural course of the endofibrosis of athletes is unknown, although stenosis, revealed by intermittent claudication is usually observed. Only a few cases of dissection and no aneurysmal degeneration have been described before.
Flow limitations in the iliac arteries of endurance athletes during exercise were previously ascribed solely to intravascular lesions. We postulate that functional kinking of the arteries can also result in flow limitations. However, the diagnostic tools in routine practice are not effective in diagnosing such flow limitations in a substantial proportion of athletes, mainly because these diagnostic tools do not measure in the provocative situations. Ninety-two symptomatic legs in 80 endurance athletes were examined with newly developed, sports-specific vascular tests. Thirty-five asymptomatic cyclists matched for working capacity served as the control subjects. Legs were classified as vascular or non-vascular following a decision algorithm, based upon the results of these diagnostic tests, excluding orthopaedic causes by the effects of specific treatment. Independently of this clinical classification, an alternative method was applied to find stable characteristics in the total patient group using factor analysis. This characterisation was based on scores on 14 test variables deriving from diagnostic tests that were not used in the decision algorithm, thus avoiding dependency between the clinical categorisation and the statistical categorisation. The hypothesis was that these characteristics were sufficiently sensitive to classify patients with vascular and non-vascular complaints. If so, these characteristics should correspond with the one derived from the decision algorithm. Following the decision algorithm, 58 legs (63%) were classified as vascular, 29 (32%) as non-vascular and 5 (5%) as inconclusive. The latter were considered non-vascular. In a substantial proportion of the vascular patients, kinking of the iliac arteries was identified as the major cause of flow limitation. The characteristics derived from factor analysis proved to classify 87% in agreement with the decision algorithm (kappa 0.56). The agreement is sufficient for validation of the clinical classification. The algorithm can therefore be applied in clinical situations to diagnose endurance athletes with flow limitations due to both intravascular lesions and kinking of the arteries.