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Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)00878-2
10.1016/j.ijscr.2024.110097
110097
Case Report
Aortic stenting of coral-reef aorta for claudication of possible neurogenic or ischemic origin – A case report
Foo Chek Hao Joshua joshuafooch@gmail.com
a⁎
Patel Ankur b
Peng Nick Ng Zhi c
a Department of General Surgery, Singapore General Hospital, Singapore
b Department of Vascular and Interventional Radiology, Singapore General Hospital, Singapore
c Department of Vascular Surgery, Singapore General Hospital, Singapore
⁎ Corresponding author. joshuafooch@gmail.com
30 7 2024
10 2024
30 7 2024
123 11009729 7 2023
21 7 2024
26 7 2024
© 2024 Published by Elsevier Ltd on behalf of IJS Publishing Group Limited.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction and importance

Coral-reef type aortic occlusions are uncommon conditions that can result in intermittent claudication. Many claudicants also have concomitant neurogenic aetiologies and revascularization alone may not be beneficial. These cases can prove to be a diagnostic challenge.

Case presentation

We present a case of worsening bilateral calf claudication in a patient with previously asymptomatic coral-reef type aorta, anxiety, and spinal stenosis presents. Investigations were unable to differentiate between a neurogenic and vascular cause. The patient opted for aortic stenting before spinal surgery, after extensive discussion.

Initial attempts at crossing the occluded segments were unsuccessful and the patient was offered the option to either resume conservative therapy, perform an open axillo-bifemoral bypass or repeat aortic stenting. After discussion, a repeat aortic stenting was performed. This time, the stenting attempt was successful, with completion angiogram showing brisk antegrade flow and strong distal pulses returned. Post-surgery, the patient's symptoms improved vastly.

Clinical discussion

Differentiation of vascular claudication from neurogenic claudication is diagnostically challenging. Decision to treat the aorta or the spinal issue first depend on the patient's constellation of symptoms.

Conclusion

Endovascular aortic stenting is well accepted with good results and lower morbidity than open surgery. A repeat attempt is always a possible option.

Highlights

• A 69-year-old with anxiety, significant spinal stenosis and a "coral-reef" type heavily calcified aorta presents with worsening bilateral calf claudication.

• The treatment of claudication can be challenging when more than one aetiology exists. Deciding which is more likely requires experience.

• Despite an unsuccessful first attempt, the eventual repeat endovascular stenting was successfully performed

• Endovascular aortic stenting is now well accepted with fairly good results and lower morbidity than open surgery. A repeat attempt is always a possible option.

Keywords

Coral-reef aorta
Aortic stenting
Claudication
Vascular
Case report
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pmc1 Introduction

Clinical presentations of aortoiliac occlusive disease vary depending on involvement of aortic branches and extent of occlusion [1]. These include intermittent claudication, rest pain, gangrene of the lower extremities, abdominal angina, and hypertension from renal artery stenosis. Coral reef aorta is a rare disease where rock-hard irregular calcified atheroma forms in the lumen of the visceral aorta, leading to severe aortoiliac occlusive disease, ischemic events involving the lower limbs or viscera, and can significantly limit the ability to perform intravascular interventions [2,3]. The presentation of such a patient with intermittent claudication who also has significant degenerative lumbar spondylosis with spinal stenosis and confounded by anxiety can prove to be both a diagnostic and management challenge.

We present a patient with previously asymptomatic infrarenal coral reef aorta with bilateral lower limb claudication who was initially treated for neurogenic pain but did not respond to treatment until vascular intervention was offered. We highlight the diagnostic challenges faced and discuss the treatment options available in patients with symptomatic infrarenal coral reef type aorta. The following case was presented in accordance with the SCARE criteria [4].

2 Case report

A 69-year-old woman with infrarenal aortic subtotal occlusion, significant degenerative lumbar spondylosis with spinal stenosis, chronic vertebral compression fractures, and mixed anxiety depressive disorder presented in 2021 with worsening intermittent claudication of 6 months' duration.

Five years prior in 2017, the patient was incidentally found to have a 1.4 cm long infrarenal segment stenosis with marked atherosclerosis of the aorta on computerized tomography (CT). Follow up CT aortograms in 2020 noted severe infrarenal abdominal aortic stenosis and a “coral reef” aorta. The patient was asymptomatic back then and was treated conservatively. She had also been seeing a psychologist for mixed anxiety depressive disorder.

In 2021, the patient developed severe worsening lower back pain over a year with radiating, “shooting pain” down her thighs. The patient also reported a new onset bilateral lower limb claudication with a short claudication distance of 50 steps. Magnetic Resonance Imaging (MRI) showed right exit foraminal stenosis from posterior indentation of the thecal sac due to ligamentum flavum hypertrophy, although no significant cord compression or oedema was seen. Transcranial magnetic stimulation was suggestive of corticospinal pathway dysfunction of the spinal cord.

The initial clinical impression was that of a neurogenic etiology and the patient was treated conservatively. Nevertheless, a repeat CT aortogram performed due to weak femoral pulses showed extensive atherosclerosis of the aortoiliofemoral region, with subtotal occlusion of the infrarenal aortic segment and severe stenoses of the bilateral iliac arteries, was suggestive of an element of ischemic pain (Fig. 1A, B). Toe pressures measured 70 mmHg. Endovascular aortic stenting was offered in view of the out-of-proportion distressing symptoms. After extensive discussion in a multi-disciplinary team, patient agreed to aortic stenting before spinal surgery. However, guidewires were unable to be advanced across the heavily calcified occluded aortic segments and the stenting attempt was unsuccessful (Fig. 1C).Fig. 1 A(top left) & B (top right): CT Aortogram showing extensive calcifications.

C (bottom left): Diagnostic angiography during first attempt at crossing occlusion.

D (bottom right): Poor distal runoff segment of the aorta and iliac arteries.

Fig. 1

Post procedure, further workup and a neurologist's opinion was sought to further assess the diagnostic uncertainty. Nerve conduction studies showed bilateral peroneal sensory neuropathy. Although the neurologist's impression was that of neurogenic pain, the patient did not respond to conservative management involving intensive physiotherapy and a trial of gabapentin. The patient was also considered for a level T10-T11 decompression laminectomy, however, she continued to experience worsening pain during this period. Therefore, further vascular intervention options were discussed to address any underlying ischemic element that may have been causing the pain.

These options included either a repeat attempt at endovascular aortic stenting, or open surgery with insertion of an axillo-bifemoral bypass graft. An aorto-bifemoral bypass was discouraged due to the high level of technical difficulty to achieve proximal and distal control when clamping the patient's heavily calcified aorta. Prior to intervention, it was made clear that these vascular interventions may not guarantee pain resolution in view of the diagnostic ambiguity. Ultimately, the patient was still keen for repeat endovascular aortic stenting to minimize surgical risks associated with major open surgery.

Arterial punctures were made via the left radial artery and left common femoral artery, and the occluded aortic segment was delineated with angiography via pigtail catheters. The stenosis was crossed intra-luminally from above using the 0.014" Winn 200 T and CXI support catheter. Serial dilation was achieved with the 6 × 80 mm Jade balloon, 10 × 80 mm Dorado balloon and 12 × 40 mm Athletis balloon. The 12 × 59 mm Bently BeGraft was then deployed over the stenosis and post-dilated with the 14 × 60 mm Atlas balloon (Fig. 2). Repeat angiogram post procedure showed brisk antegrade flow, and good pulses were felt at the left common femoral artery.Fig. 2 A (top left): occlusion successfully crossed intra-luminally with guidewire

B (top right): Serial dilatation with Dorado and Athletis balloons

C (bottom left) & D (bottom right): Completion angiogram showing brisk antegrade flow across the previously occluded segment.

Fig. 2

The patient subsequently reported gradual and complete resolution of her symptoms. She was discharged home uneventfully and planned for subsequent follow ups with interval doppler ultrasound scans of the aorta.

3 Discussion

3.1 Determining the cause of claudication

Differentiation of vascular claudication from neurogenic claudication has always been a diagnostic challenge. Despite the advances in noninvasive imaging technology, not all patients with peripheral vascular disease as suggested by Doppler ultrasound and arterial brachial index (ABI) measurement, report claudication symptoms. Similarly, positive lumbar spinal stenosis seen on MRI or CT may be present in asymptomatic individuals, hence poorly correlating with lower limb symptoms [5].

This diagnostic challenge has been well documented in literature. A similar case series reported by Dodge et al. described nine cases with concurrent lumbar spinal stenosis and peripheral vascular disease with claudication symptoms that were all initially treated with decompression laminectomy; only seven out of the nine patients had resolution or improvement of symptoms [6]. Nadeau et al. also described the classic symptoms for differentiation between neurogenic and vascular claudication as independently weakly valid, although certain symptom constellations yield stronger associations [7]. Unfortunately, our patient did not present with the specific constellation mentioned.

The diagnostic uncertainty and decision for intervention in this case could have been exacerbated by the patient's mixed anxiety depressive disorder. Smolderen et al. reported higher rates of exertional leg symptoms (claudication, atypical leg symptoms, rest pain) in patients with anxiety or impaired mood compared to those without [8]. Hence, the decision to treat the aorta or the spinal issue first will depend heavily on the patient's constellation of symptoms, but it is a difficult decision, nonetheless.

3.2 Treatment options for coral reef aortas

Coral-reef type aortas are uncommon with a previously reported prevalence rate of approximately 0.6 %, and the average age of presentation about 50 years old [9]. The treatment of choice for symptomatic coral reef aorta was traditionally open surgical transaortic thromboendarterectomy, whereby the affected aortic segment in opened longitudinally and the intraaortic calcifications removed [2,10]. In recent times, there have been multiple reports of the successful use of endovascular therapies (EVT) [11]. Holfeld et al. described symptom resolution with no recurrence 6 months after undergoing transfemoral endovascular stent-graft placement in two cases of symptomatic coral reef aorta [12]. EVT reduces the risks of major open surgery and may be more appropriate in patients with multiple comorbidities compared to conventional surgical interventions.

However, endovascular aortic stenting requires an adequate landing zone, especially with use of graft stents, and care must be taken to avoid covering of visceral arteries [13]. Aortic stenting may also be technically challenging as severe intraluminal calcifications may significantly limit the ability to perform intravascular interventions, as with our case. Although due to its minimally invasive nature, EVTs can be repeated on a different day, using a different wire, approach and operator should the patient's renal function allow.

Transaortic thromboendarterectomy is a major open surgery with significant morbidity and high technical difficulty, especially when attempting to fully clamp the patient's heavily calcified aorta before endarterectomy. Aortobifemoral or axillobifemoral bypass grafting may be an alternative, less invasive treatment modality [14]. Bypass can be offered for cases with substantial extension along the aorta or renal arteries. However, there are early and late graft complication risks that could result in downstream malperfusion. Primary patency rates were reported to be between 50 % and 80 % after three years [15]. Extensive anticoagulation will also be required which contain inherent anticoagulation related adverse events.

In our case, repeat stenting using different wires and approach, performed by a different operator was offered after the first unsuccessful attempt, in view of the morbidities associated with open surgery. Had repeat stenting not been successful, an axillobifemoral bypass graft would have been offered instead of endarterectomy or aortobifemoral bypass due to the rim of thick calcifications in the aortic lumen, and technical difficulty to anastomose the proximal graft. There would also have been additional visceral cardiac and hernia risk as well.

4 Conclusion

In conclusion, we report the diagnostic challenge faced with a case of worsening intermittent claudication in a patient with possible neurogenic, ischemic, or psychogenic origin. Repeat aortic stenting can be offered as an alternative, less invasive treatment option despite unsuccessful initial attempts.

Consent

We confirm that written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical approval

Ethical approval for this study is not required, in this case report of a singular case in our instituition. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Funding

Nil.

Author contribution

Dr Chek Hao Joshua Foo – Author

Dr Ankur Patel – contributed to patient care, provided patient for the study

Dr Nick Ng Zhi Peng- contributed to patient care, provided patient for the study, Author

Guarantor

Guarantor: Dr Nick Ng Zhi Peng

Research registration number

NA.

Conflict of interest statement

Nil.
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