J Vasc Access 2014 ; 15 (5): 427- 430 CLINICAL CASE

DOI: 10.5301/jva.5000279

Acute limb ischaemia due to focal brachial artery aneurysms

complicating brachiocephalic arteriovenous fistula ligation:
two recent case reports

Sandeep S. Bahia1, Francesca Tomei2, Baris A. Ozdemir1, Eric S. Chemla1

1
St George’s Vascular Institute, London - UK
2
Department of Vascular Surgery, University of Pisa, Pisa - Italy

ABSTRACT
Introduction: True brachial artery aneurysms are rare, typically occurring secondary to trauma. In this report, we describe
two recent cases of patients who presented acutely with upper limb ischaemia due to brachial artery aneurysms. Both
patients presented many years after brachiocephalic arteriovenous (AV) fistula ligation in the ipsilateral limb.
Report: Two male patients, aged 60 and 63 years, respectively, were seen acutely with symptoms of upper limb ischaemia.
They had both undergone ligation of AV fistulae many years earlier having received functioning transplants. Subsequently,
both patients were found to have true brachial artery aneurysms, which were bypassed in both instances using great saphe-
nous vein grafts.
Discussion: Patients undergoing ligation of AV fistulae should receive interval surveillance imaging to detect potential aneu-
rysmal dilatation of upper limb vessels. Little is known about the incidence of aneurysm formation after AV fistula ligation;
given the increasing number of patients undergoing dialysis, and hence the burgeoning number of patients who may receive
transplants, it is important that upper limb ischaemia is pre-empted by appropriate follow-up.

Key words: Aneurysm, Arterial, Brachial, Fistula, Ischaemia, Surveillance

Accepted: June 4, 2014

INTRODUCTION A true aneurysm is typically taken to mean “a per-

manent, focal dilation of an artery having at least a
The diameter of a normal brachial artery varies from 50% increase in diameter compared to the expected
3.5 to 4.3 mm for women and from 4.1 to 4.8 mm for normal diameter of the artery in question.” Upper ex-
men. Brachial artery aneurysms (BAAs) have a prevalence tremity aneurysms as a whole are rare; when compared
of 0.5% (1) and are most commonly false aneurysms sec- with other vessels in the body, they represent 0.6% of
ondary to trauma or previous arteriovenous (AV) fistulae total aneurysm repairs (12), with the commonest clini-
(2); however, true BAAs can be either focal or more gen- cal presentation being distal thromboembolism, rather
eralized. than rupture.
A PubMed literature search aimed at identifying exist- Generally, infectious, post-traumatic or iatrogenic ae-
ing literature relating to true upper limb aneurysms (ULAs) tiology is recognized as the most common cause of false
was conducted returning 191 papers of which only 60 and true brachial aneurysms. Studies have previously
were relevant. The vast majority of these (52/60) were case identified a correlation between brachial artery diameter
reports (1, 3, 4), with the remainder reports of aneurysms and the length of time that a fistula has been in situ (13);
related to previous renal access work (5, 6) or other dis- however, very few cases have been described of true BAAs
ease processes such as arteritis (7) or haemophilia (8). Of after ligation of a fistula.
the case reports documenting true ULA, there is nearly A recent review article conducted a literature review
always a history of repeated blunt trauma (9). Cases of to identify reported cases of BAA formation complicating
late aneurysm formation have been described in patients AVF ligation following renal transplantation; the paper re-
who have had AV fistulae (AVFs) ligated but remain rare vealed that very few cases are formally documented in the
(10, 11). available literature (10).

© 2014 Wichtig Publishing - ISSN 1129-7298 427

Surveillance is needed after AVF ligation to detect aneurysm formation

It is likely that the aetiology of true aneurysms is minal thrombus. Thrombectomy was carried out to assess
related to the release of endothelium-derived factors. Fur- inflow, which was good, and the decision made to ligate
thermore, the use of corticosteroids in transplant patients the native brachial artery and carry out an axillo-brachial
has been demonstrated to promote both the development artery bypass using an interposition reversed great saphe-
and rupture of aneurysms (13). nous vein (GSV) graft. At the end of the procedure, there
We describe two cases of renal transplant patients, was a palpable radial pulse, with a warm well-perfused
who had undergone AVF ligation in the ipsilateral limb, hand.
presenting with acute upper limb ischaemia complicating Histology and microbiology tests of the thrombus did
a previously undiagnosed focal BAA. not reveal signs of infection or any other pathological his-
tology.
Post-procedure investigations [echocardiogram, total
Case report 1 body computed tomography (CT) angiogram to exclude
any other aneurysms and haematological work up given a
A 60-year-old man was admitted with a 3-week history family history of Factor V Leiden] were all grossly normal.
of symptoms of ischaemia in his left arm. The patient recovered uneventfully with complete resolu-
His past medical history consisted of renal failure tion of his symptoms.
secondary to mesangio-capillary glomerulonephritis; as
a consequence, at the age of 31, he underwent fashion-
ing of a left brachio-cephalic fistula through which he Case report 2
received haemodialysis for 1 year. He received two non-
living donor kidney transplants, the first aged 32 years A 63-year-old man was admitted to the same
who failed as a complication of treatment for a semi- vascular unit with intermittent pain in the left hand,
noma at the age of 34 years, and the second 3 years after typically worse on mobilising the limb, suggestive of
this. ischaemia.
He was maintained on immunosuppressive therapy The patient had undergone renal transplantation at
consisting of azathioprine and prednisolone. Sixteen the age of 41, having developed renal failure secondary
years after the second transplant, aged 53 years, the to congenital renal hypertensive disease during his 20’s.
patient underwent elective ligation of the left brachio- This fistula was ligated and the radio-ulnar bifurcation
cephalic fistula; this was carried out as the patient no refashioned using PTFE 14 years ago as he developed a
longer required the fistula and had developed an un- large pseudoaneurysm at the anastomosis. Subsequently,
sightly, grossly dilated fistula, in the absence of any his transplant failed and he now dialyses through a right
symptoms. The operation note at the time states that the brachiocephalic AVF. During the 17 years that his kidney
brachial artery was repaired “using 2 layers of running transplant was functioning, he was on immunosuppres-
5.0 Prolene,” but makes no reference to any dilatation sive and corticosteroid therapy.
of the vessel. Other relevant risk factors in his previous history con-
Seven years later after the ligation, he presented sisted of hypertension, hypercholesterolemia, coronary ar-
with the aforementioned ischaemic upper limb. At pre- tery disease that required a coronary artery bypass grafting
sentation, his sensation and motor function were nor- (2011), and peripheral vascular disease that required an
mal; however, he had impalpable pulses in the limb. An aorto-bifemoral bypass (2009).
arterial duplex was obtained that showed complete oc- The upper limb symptoms that he presented with had
clusion of the axillary and brachial arteries, with no flow been present for 2 months but were now associated with
in the ulnar artery and monophasic, low velocity flow in pallor, cold fingertips and paraesthesia. An arterial du-
the radial artery. A diagnostic angiogram was not carried plex showed a 30 mm BAA with an occluded prosthetic
out at this point to avoid nephrotoxic contrast exposure radio-ulnar bifurcation graft. The patient was admitted,
and because the clinical presentation and the ultra- placed on intravenous heparin and scheduled for urgent
sound scan appeared to sufficiently present a diagnosis surgery.
of limb ischaemia complicating the previous interven- He underwent an exclusion bypass to the ulnar artery
tions that the patient had undergone. He was placed on with reimplantation of the radial artery using a reversed
intravenous heparin infusion and scheduled for urgent GSV graft. There was a palpable radial and ulnar pulse at
surgery. the end of the procedure.
The patient underwent exploration of the brachial Histology and microbiology carried out on the
artery in the antecubital fossa and in the upper arm; it thrombus showed no evidence of infection or pathologi-
was immediately apparent that there was a grossly aneu- cal histology. The patient recovered uneventfully with
rysmal brachial artery, measuring 2.5-3 cm, which had complete resolution of his symptoms and was discharged
become occluded secondary to a large volume of intralu- home.

428 © 2014 Wichtig Publishing - ISSN 1129-7298

Bahia et al

DISCUSSION more proactive in identifying patients who may develop

them. Five of these (5/13, 38.5%) developed after ligation
We describe two recent cases of late, true BAA forma- of the AVF at a mean of 3.6 years (range 2-6 years) (11).
tion after brachiocephalic AVF ligation. True BAAs are rare, From a technical perspective, it is important when ligat-
and a review of the literature shows very few documented ing such AVF to ensure that a long stump is not left behind,
cases in the context of a prior ligation of an AVF (10, 11). even if this means that an end-to-end resection anastomosis
Prompt treatment avoids the potentially catastrophic se- is necessary; this reduces the potential for aneurysmal dila-
quelae of upper limb ischaemia and amputation; the op- tation around the ligation site. Interestingly, neither of these
timal intervention is bypass with preferably vein or, failing cases had such a stump remaining, indicating that technical
that, prosthetic graft. issues at the time of ligation are not to blame.
Modality of imaging in the preoperative assessment Given that this group of patients are typically high-risk
of these patients is often complicated by a desire to avoid for arterial disease and the development of atherosclerotic
nephrotoxic contrast. Prospective studies have demonstrat- disease, and given that our recent experience suggests that
ed that, certainly in the context of lower limb ischaemia, they may be more likely to develop arterial aneurysms after
duplex imaging is as good as CT angiography in delineat- ligation of a previous fistula, it is our feeling that all patients
ing anatomy (14). However, as seen in the first case here, who undergo AVF ligation warrant surveillance imaging to
this can be misleading, as it may not be possible to see the exclude aneurysmal dilatation of the proximal upper limb
overall vessel diameter. In most vascular units in the UK, arteries. With the increasing availability of USS for surveil-
experienced sonographers now conduct the vast majority lance, and given that it would appear that brachiocephalic
of vascular imaging using ultrasound scan (USS) primarily, AVFs carry the highest risk of resulting in BAA after ligation,
with CT used to supplement imaging when an abnormality we would suggest that those patients undergoing ligation of
is suspected. For more complex situations such as those de- a brachiocephalic AVF should undergo annual arterial du-
scribed here, it may be the case that a more detailed form of plex USS surveillance. Transplant teams who ligate the AVF
imaging such as MRA or CTA might be appropriate with ad- after successful transplantation would best coordinate this
equate renal physician support to minimize nephrotoxicity. surveillance; this would avoid patients being lost to follow-
It is likely that the true number of patients who de- up and pre-empt the potentially catastrophic sequelae of
velop BAA after ligation of AVF is much higher; the fact acute upper limb ischaemia.
that aneurysms are typically asymptomatic until an acute
event occurs may be a factor in the under-reporting of this Financial support: The authors have no financial disclosures to
phenomenon. One also has to bear in mind that, histori- make.
cally, a relatively small number of patients may have sur-
vived long enough for this to be a factor; the mean time to Conflict of interest: The authors have no conflict of interest.
presentation in the two cases described here is 10.5 years.
Previously published work from our unit retrospective-
Address for correspondence:
ly examined patient data from the period January 2006– Sandeep Singh Bahia
July 2009; this identified 13 true BAAs from a pool of 288 St George’s Vascular Institute
patients who had previously undergone autogenous bra- Blackshaw Road
chiocephalic AVF fashioned (4.5%); this is in keeping with Tooting
AAA = Abdominal aortic aneurysm prevalence in the pop- London, SW17 0QT, UK
ulation as a whole, suggesting perhaps that we need to be [email protected]

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