International Journal of Surgery Case Reports 95 (2022) 107253

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International Journal of Surgery Case Reports

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Case report

A distinguished case of a spontaneously dissecting Left Common Iliac

Artery Aneurysm, associated with an Elastinopathy, in a healthy 36-year-­
old male, successfully treated with open surgical repair - A Case Report
Omar Hariri a, b, Omar Al Laham a, b, *, Ammar Mohammad c
a
Department of Surgery, Al-Mouwasat University Hospital, Mazzah, Damascus, Syria
b
Department of Surgery, Al Assad University Hospital, April 17th St. Kafar Sousah, Damascus, Syria
c
Department of Vascular Surgery, Al Assad University Hospital, April 17th St. Kafar Sousah, Damascus, Syria

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction and importance: Elastin is a major protein of the Extracellular Matrix (ECM), essential in providing
Case Report elasticity to the vascular wall, which enables blood vessels to reversibly expand and contract.
Elastinopathies Several inherited or acquired etiologies, such as elastinopathies and fibrillinopathies negatively impact the
Aneurysms
objective of the Extracellular Matrix via compromising the Elastin fibers in the Cardiovascular System (CVS) and
Spontaneously dissecting Iliac Artery
Aneurysm
the skin.
Vascular Surgery emergency Such compromises will have devastating ramifications through the increase in vascular wall stiffness and the
Aorto-bi-Femoral Bypass inability to properly dissipate energy. This impact on the vascular wall will contribute to the development of
arterial aneurysms and dissections.
Case presentation: Our case is of a 36-year-old previously healthy male patient who presented with an acute onset
of left lower limb pain associated with cold sensation two days prior to admission. Radiology demonstrated a
dissecting aneurysm in the left Common Iliac Artery.
Clinical discussion: The patient was surgically treated with an Aorto-bi-Femoral Bypass (ABFB).
Histopathological analysis of the excised aortic and iliac specimens revealed no Elastin fibers in the vessel walls.
Conclusion: Isolated Iliac aneurysms are a rare entity and are challenging to diagnose preoperatively. In our case,
there were no risk factors whatsoever and the patient's history - including family history - was negative. It is vital
to establish preoperative diagnostic approaches in such cases and keep them in mind so that we can diminish the
morbidity and mortality resultants from the complications.

1. Introduction witnessed in older males with a history of hypertension, smoking, or less

commonly due to iatrogenic injury, trauma, arteritis, connective tissue
Aneurysms are defined as any pathological increase of more than pathologies, or infections [6–10]. Classically, they are asymptomatic,
50% of the original arterial diameter. Isolated Iliac Artery Aneurysms thus, problematic to diagnose preoperatively during physical examina­
(IAAs) are exceptionally rare, comprising less than 2% of all aneurysmal tion unless they reached a large size evident on palpation. Nonetheless,
pathologies with an incidence rate of merely 8–11% in patients older preoperative suspicion can be originated if there was a compromise of
than 65 years of age [1,2]. We consider an Iliac arterial diameter of more nearby vital structures (i.e., the urinary system, bowel loops, nerve
than (1.8 cm) in males or more than (1.5 cm) in females to constitute the roots, pelvic veins), as a complication of thrombus formation, rupture,
diagnosis of an aneurysm [1,3]. We triage a patient with an Iliac Artery or dissection [9]. Patient presentation varies, this can be depicted by
diameter exceeding (3.5 cm) [4] or an expansion rate of more than (1 several ill-defined symptoms, the commonest of which is sudden
cm) per year, a candidate for intervention [5]. This is conventionally abdominal, groin, or flank pain. Moreover, patients report intermittent

Abbreviations: ECM, Extracellular Matrix; CVS, Cardiovascular System; ABFB, Aorto-bi-Femoral Bypass; IAAs, Iliac Artery Aneurysms; CRP, C-Reactive Protein;
ESR, Erythrocyte Sedimentation Rate; DUS, Doppler Ultrasound; CTA, Computed Tomography Angiography; AAAs, Abdominal Aortic Aneurysms; CT, Computed
Tomography; SMCs, Smooth Muscle Cells; MRI, Magnetic Resonance Imaging.
* Corresponding author at: Department of Surgery, Al-Mouwasat University Hospital, Mazzah, Damascus, Syria.
E-mail addresses: omar.hariri1992@[Link] (O. Hariri), 3omar92@[Link] (O. Al Laham), ammarmhd@[Link] (A. Mohammad).

[Link]
Received 29 April 2022; Received in revised form 25 May 2022; Accepted 28 May 2022
Available online 30 May 2022
2210-2612/© 2022 The Author(s). Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
([Link]
O. Hariri et al. International Journal of Surgery Case Reports 95 (2022) 107253

claudication or chronic vague lower limb discomfort [9]. This leads to concentrated in left groin, the limb's temperature was spontaneously
misdiagnoses, which in turn, raise the rates of morbidity and mortality. restored. However, the pain was unresponsive to over-the-counter an­
The work has been reported in line with the SCARE criteria and the algesics. It was aggravated by ambulation and partially relieved by lying
revised 2020 SCARE guidelines [30]. down. He reported a history of lacrimation when subjected to light
sources. No intermittent claudication, fever, vascular ulceration, or skin
2. Presentation of case changes were stated. Moreover, no genitourinary symptoms or alter­
nation of bowel habits were reported. The patient is an ex-smoker with a
2.1. Patient information 3-pack-year smoking history. He isn't an alcoholic. His BMI is 19 kg/m2.
His height is 1.5 m. Surgical history involved an open Appendectomy 20
We present the case of a previously healthy 36-year-old Middle years ago and two exploratory laparotomies for adhesiolysis. Familial,
Eastern male patient, who presented to the Vascular Surgery clinic at our drug, and allergic histories were all negative.
tertiary university hospital with symptoms commencing 10 days prior to
admission. His story instigated as an acute onset of unspecific left lower
2.2. Clinical findings
limb pain. It was sudden, persistent, scaling 4 out of 10 according to the
patient, and associated with numbness, paresthesia, and coldness of the
Vital signs were normal. Upon inspection, no skin changes were
affected lower limb, which lasted for two days. Afterwards, the pain was
noted -including ulceration, pallor, ecchymosis, or reticular spotting-

Fig. 1. A–B: Computed Tomography Angiography (CTA) coronal view yielded; Terminal abdominal aortic dilation measuring (3.0 cm), and an isolated dissected left
Common Iliac Artery aneurysm, estimated to measure (5.5 × 4.5 cm). No calcifications were noted.
C: Computed Tomography Angiography (CTA) three-dimensional view yielded; Terminal abdominal aortic dilation measuring (3.0 cm), and an isolated dissected left
Common Iliac Artery aneurysm, estimated to measure (5.5 × 4.5 cm).
D: Computed Tomography Angiography (CTA) sagittal view yielded; Terminal abdominal aortic dilation measuring (3.0 cm), and an isolated dissected left Common
Iliac Artery aneurysm, estimated to measure (5.5 × 4.5 cm).

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O. Hariri et al. International Journal of Surgery Case Reports 95 (2022) 107253

Upon palpation, pulse was vividly palpable along the entire arterial axis undiagnosed connective tissue such as the vascular variant of Ehlers-
of the left lower limb. No palpable masses in the abdomen, groin, or Dalnos Syndrome or an overall Elastinopathy.
popliteal fossa were detected. Upon auscultation, a notable thrill was To evade the unpredictable intraoperative complications, an ABFB
heard over the path of the left Common Femoral Artery. No other thrills was deemed to be the therapeutic intervention. Based on the patient's
were auscultated over the rest of the arterial axis. prior surgical history, a classical midline abdominal incision was cir­
cumvented, and a pararectal retroperitoneal incision was done. The
2.3. Laboratory investigations intraoperative findings confirmed the preoperative radiological
analysis.
C-Reactive Protein (CRP) was normal and was 0.1 mg/dl. Erythro­ As suspected prior to surgery, the arterial walls were abnormally
cyte Sedimentation Rate (ESR) was normal and was 12 mm/h in the 1st stretching and were friable during the anastomotic phase of the opera­
hour. The remaining laboratory panel was within normal ranges. tion, we covered the aortic anastomosis with a tube of Dacron to mini­
mize the risk of developing aneurysms and to protect the anastomosis
from rupture. The Inferior Mesenteric Artery, the terminal Aorta, the
2.4. Diagnostic assessment
External and Left Internal Iliac Arteries were ligated. An ABFB was done
with a Dacron (16 × 8 cm) prosthesis (Fig. 2A–B).
Doppler Ultrasound (DUS) revealed a dissecting left Common Iliac
A full thickness aortic wall biopsies from the right and left Common
Artery aneurysm spanning into the left External Iliac Artery, with a vi­
Iliac Arteries and the abdominal Aorta, with proper length was taken to
sual flap in it. It measured approximately (4 × 5 cm) with wall throm­
confirm the suspected etiology behind the diagnosis. Histopathological
bosis seen within it. The remainder of the arterial axis was normal. Upon
analysis revealed dilation and an organized thrombus formation.
admission, a Computed Tomography Angiography (CTA) was done and
Furthermore, no elastic fibers were noted (Fig. 3A–D). The patient had
yielded; terminal abdominal aortic dilation measuring (3.0 cm), and an
an uneventful postoperative recovery and was discharged within 5 days
isolated dissected left common Iliac Artery Aneurysm, estimated to
of surgery. He was provided with instructions which ameliorate his re­
measure (5.5 × 4.5 cm). No calcifications were noted (Fig. 1A–D).
covery (i.e., regular sterile wound dressings, analgesics, and a pre­
Initial management included keeping the patient in a nil-per-mouth
scription of postoperative antibiotics). A follow-up protocol was
status, establishing intravenous access, prophylactic antibiotics, com­
assigned in the outpatient settings for 6 months now. He has had regular
plete blood panel, blood sampling and crossmatch for surgical prepa­
appointments at the Vascular Surgery clinic to undergo surgical exam­
ration. Noteworthy challenges were the unavailability of a laparoscopic
ination and DUS imaging.
machine in the Emergency Department at the time of surgery and the
inability to perform genotyping for the patient due to his low socio­
3. Discussion
economic status and unavailability of the required materials in the
accessible labs.
Ad Hoc Committee depicted in their report to the Society for
Vascular Surgery in 1991 that what constitutes an arterial aneurysm is
2.5. Therapeutic intervention when an artery suffers from 50% increase in its original diameter as a
minimum [11] The most prevalent occurrence site for extracranial
Based on the clinical preoperative assessment which was comprised arterial aneurysms is in the infrarenal aortic segments [12–14]
of the following elements: A previously healthy male with short stature, Furthermore, Abdominal Aortic Aneurysms (AAAs) are estimated to be 9
low BMI, young age, negative familial and risk factor histories, aortic times more rampant than thoracic aortic aneurysms [15]. It is settled
ectasia, and an isolated dissecting Common Iliac Artery aneurysm that merely 5% of males and 1.7% of females who are older than 65
(Arterial wall dilation in more than one segment), a rheumatologic years of age will develop an infrarenal aortic segment aneurysm with a
consultation was warranted. Surgery was accomplished at out university diameter larger than (3 cm). Nevertheless, it's projected that there's a 6%
hospital by a Vascular Surgery consultant with 15 years of Vascular increase in the chance of developing an aortic aneurysm for every sub­
Surgery experience. It was undergone under general anesthesia with no sequent decade after the previously mentioned age [16].
complications. It concluded that this case could be a result of an

Fig. 2. A: Intraoperative image where the black arrow depicts the dissected aneurysmal Iliac Artery lumen (Arrow).
B: Intraoperative image depicting the ABFB which was done with a Dacron (16 × 8 cm) prosthesis.

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O. Hariri et al. International Journal of Surgery Case Reports 95 (2022) 107253

Fig. 3. A: Histopathological analysis revealed arterial

dilation and an organized thrombus formation.
B: Histopathological analysis of the right Common
Iliac Artery revealing the absence of Elastin from the
arterial wall.
C–D: Histopathological analysis of left Common Iliac
Artery revealing the dissection and the absence of
Elastin from the arterial wall. Furthermore, no Elastic
fibers were found.

The most common segments affected by the pathophysiology of component and directed towards the elastic lamella. We must emphasize
aneurysmal degeneration are in descending order; the common iliacs the recurrent failure of clinical physical examination in discovering IAAs
(70–90%), the internal iliacs (10–30%), or those two simultaneously. In [20].
contrast, an External Iliac Artery aneurysm is ponderously rare in We reliably resort to radiological analysis due to its proven sensi­
occurrence [3,17]. tivity and specificity in identifying such aneurysms and in turn, plan for
Conventionally, IAAs are stumbled-upon incidentally preoperatively surgical interventions. For example, Doppler Ultrasound in the hand of
whilst clinical screening for a AAA, especially when a patient undergoes an expert user constitutes the corner stone for screening and diagnosis of
ultrasonography or an abdominal/pelvis Computed Tomography (CT) aneurysms. Moreover, CT and Magnetic Resonance Imaging (MRI) are
scan to diagnose other suspected pathologies [1,3]. This is classically the gold standard for depicting a detailed anatomical image of the
observed in older males with a history of hypertension, smoking, or less affected arteries, hence, providing an enhanced accuracy in diagnosing
commonly due to iatrogenic injury, trauma, arteritis, connective tissue and documenting such aneurysms. However, planning surgical in­
pathologies, or infections [6–10]. terventions can be challenging and requires an extensive degree of im­
Traditionally, they are asymptomatic, thus, problematic to diagnose aging analysis. This is provided by CT arteriography because of its high-
preoperatively during physical examination unless they reached a large resolution imaging output. Finally, with the aid of radiology software,
size evident on palpation. Nonetheless, preoperative suspicion could be we depict 3-D postprocessing images to fully visualize the vascular tree
present if there was a compromise of nearby vital structures (i.e., the and the affected arterial segments [21].
urinary system, bowel loops, nerve roots, pelvic veins), as a complica­ Symptomatic IAAs warrant surgical intervention. The current
tion of thrombus formation, rupture, or dissection [9]. consensus prefers an elective repair for asymptomatic IAAs with a
The integrity and function of an arterial blood vessel walls is main­ diameter greater than (3.5 cm) in previously healthy individuals [4].
tained by key elements such as the delicate balance between the However, asymptomatic IAAs with a diameter smaller than (3 cm) are
degradation of the ECM by proteins, mechanical patency, inflammation preferred to be surveilled with regular imaging modalities [21,22].
processes, and the malfunctioning remodeling complex mechanism. This There's a considerable risk for bleeding in conventional surgical in­
all occurs in the setting of increased risk-patients, whether due to terventions. It is especially challenging because of the anatomical situ­
congenital predisposition or sporadic environmental factors [4,18]. ation of IAAs in the pelvis, in addition to the high density of pelvic veins,
When it comes to the integrity and aortic blood vessel wall patency, the coexisting atherosclerotic plaques and calcifications [23].
Elastin fibers are key components necessitated for the proper recoil and Previously healthy patients who possess unremarkable risk factors
expansive tasks of the arterial walls. Any dysfunction in those fibers can undergo a smooth and successful open surgical operation to repair IAAs.
lead to devastating effects in the affected arteries [19]. The main purpose behind this surgery is to keep a healthy distal limb
We must also highlight the vitality of Elastin fibers working blood perfusion whilst attempting to stop any more degree of arterial
conjointly with Smooth Muscle Cells (SMCs) to enable the arterial wall wall degeneration, thus dissection [20].
to correctly transmit the mechanical signals emitted by the actinomysin The open surgical technique classically involves a midline,

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O. Hariri et al. International Journal of Surgery Case Reports 95 (2022) 107253

transplant-type, or a retroperitoneal incision. This is based on the pre­ Provenance and peer review
vious surgical history of the patient, the extent of said aneurysm,
whether the pathology is uni-/bilateral, and naturally, the surgeon's Not commissioned, externally peer-reviewed.
experience.
One essential benefit of open surgical repair of IAAs is the ability to Ethical approval
maintain adequate collateral flow of blood to the pelvis via the mesen­
teric and lumbar arteries. This will in turn, considerably diminish the Institutional review board approval is not required for deidentified
risk of postoperative pelvic and/or colonic ischemia [24]. single case reports or histories based on institutional policies.
The general prevalence of perioperative complications can rise-up to
22%. This demarcates lower limb ischemia, visceral or pelvic organs Funding
ischemia, aneurysmal dissection or rupture, arterioenteric fistula
development, infection of used graft, sepsis, and surrounding organs This research did not receive any specific grant from funding
injury [4,17,25]. agencies in the public, commercial, or not-for-profit sectors.
Regarding graft patency, early and late complications are seldom
seen. Nonetheless, its primary efficacy at the 5-year mark could reach Guarantor
100% [26,27].
The ongoing scientific advancement in the field of newer generation Omar Al Laham.
bifurcated AAA grafts has morphed the surgical intervention from its
open approach to the lesser-risk endovascular approach. Endovascular Research registration number
treatment of aortoiliac aneurysms and IAAs has therefore been favored if
possible, over the classical open surgical intervention [28]. N/A.
The endovascular approach to treatment of IAAs has proven to
plunge perioperative patient complications, reduce hospital stay for CRediT authorship contribution statement
patients, decrease intraoperative blood loss, and avoid the need for
intensive care unit admissions postoperatively [29]. OA, OH: Conceptualization, resources, who wrote, original drafted,
The prevalence of ruptured IAAs at the time of initial diagnosis can edited, visualized, validated, and literature reviewed the manuscript.
reach up to 33% and this is accompanied by an imminent risk of AM: Vascular Surgery Specialist, who performed and supervised the
increased mortality. This is described by Bacharach et al. [9]. operation. Supervision, project administration, and review of the
Emergency surgical intervention is associated with an estimated manuscript.
mortality risk of 28% in the past two decades. This is compared to a OA: The corresponding author who submitted the paper for
marked mortality risk of 5% for the elective surgical repair of IAAs [4]. publication.

4. Conclusion All authors read and approved the final manuscript.

Elastin is a major protein component of the ECM. Its presence and its
collaboration with the SMCs and other ECM constituents, play an un­ Declaration of competing interest
equivocal role in the CVS and skin patency and function. Elastinopathies
are rare and result in devastating consequences such as dissecting The authors declare that they have no competing interests.
arterial aneurysms, which will in turn, lead to increased morbidity and
mortality for patients. The diagnosis of congenital Elastinopathies can Acknowledgements
further protect other asymptomatic family members by setting-up reg­
ular screening dates to avoid any possible complications. - Radiology Department at Al Assad University Hospital, Damascus,
Clinical suspicion ought to be present when presented with such Syria. For their role in radiological imaging and result interpretation.
cases so that we can perform timely interventions which limit the - Pathology Department at Al Assad University Hospital, Damascus,
complications of such pathologies. Surgical intervention remains the Syria. For their role in the histopathological analysis and interpre­
gold standard treatment modality with the preferred approach being tation of the excised specimens which helped to establish the
endovascular rather than open surgical techniques. Nonetheless, prog­ definitive diagnosis.
nosis is satisfactory, and the success depends on a multifactorial set of
elements, the most important of which is dependent on each individual
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