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Introduction
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-
story.
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.
G.F. VERALDI, M. MACRÌ, P. CRISCENTI, L. SCORSONE,
C.C. ZINGARETTI, M. GNONI, L. MEZZETTO
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.F. VERALDI, M. MACRÌ, P. CRISCENTI, L. SCORSONE,
C.C. ZINGARETTI, M. GNONI, L. MEZZETTO
G Chir Vol. 36 - n. 6 - pp. 267-271
November-December 2015
267
Department of Vascular Surgery, Polo Chirurgico “Pietro Confortini”,
University Hospital of Verona, Verona, Italy
Corresponding author: Gian Franco Veraldi,
e-mail: gianfranco.veraldi@ospedaleuniverona.it
© Copyright 2015, CIC Edizioni Internazionali, Roma
clinical practice
6 Arterial_VERALDI.qxp_- 10/02/16 15:48 Pagina 267
268
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.
AB
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).
AB
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.
269
Arterial endofibrosis in professional cyclists
AB
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.
Discussion
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).
Conclusions
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.
270
G.F. Veraldi et al.
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271
Arterial endofibrosis in professional cyclists
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