Case Report Aortoiliac Occlusive Disease Presenting as Sudden Onset Paraplegia Chien-Hung Lai,1,2 Cheng-Hsien Wang,1,2 Shih-Yun Wu,1,2 and Hong-Mo Shih,1,2 Puzi City, Chiayi, Taiwan

Thromboembolism and atherosclerotic stenosis both can cause arterial occlusion. Aortoiliac occlusive disease involving bifurcation of the aortoiliac artery induces symptoms of ischemia such as claudication and pain of buttocks and thighs, decreased bilateral femoral pulses, and impotence. Here, we describe a 58-year-old woman with a past history of atrial fibrillation and lacuna stroke with minimal right side weakness. She presented to our emergency department with sudden onset bilateral pain in the legs and paraplegia. A comprehensive examination revealed paresthesia and decreasing bilateral distal pulses. Computed tomographic imaging showed filling defects over the low abdominal aorta just above the bifurcation of the common iliac artery and bilateral femoral arteries. Acute aortic embolic occlusion was suspected. Her symptoms were resolved after emergent thrombectomy for acute limb ischemia. Physicians need to be aware of aortoiliac embolic occlusive disease which may present as acute paraplegia.

Aortoiliac occlusive disease, also known as Leriche syndrome,1 is an atherosclerotic disease involving the abdominal aorta and/or bilateral iliac artery. Patients with Leriche syndrome typically have chronic onset triad symptoms such as intermittent claudication of buttocks and thighs, decreased bilateral femoral pulses, and impotence in men. The decreased blood flow can also result in pain in the back, buttocks, and lower limbs. However, there are few reports on acute aortoiliac occlusion. Here, we report a rare presentation of acute aortoiliac

1 Department of Emergency Medicine, Chang Gung Memorial Hospital, Puzi City, Chiayi, Taiwan. 2 Chang Gung University College of Medicine, Puzi City, Chiayi, Taiwan.

Correspondence to: Hong-Mo Shih, MD, Department of Emergency Medicine, Chang Gung Memorial Hospital, Chiayi No.6, West Section, Jiapu Road, Puzi City, Chiayi County 613, Taiwan; E-mail: [email protected] Ann Vasc Surg 2014; -: 1–3 http://dx.doi.org/10.1016/j.avsg.2013.12.013 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: October 20, 2013; manuscript accepted: December 18, 2013; published online: ---.

embolic occlusive disease causing acute bilateral ischemia in the legs.

CASE REPORT A 57-year-old woman was sent to our emergency department (ED) at midnight because of sudden onset of pain and weakness in the bilateral legs. She reported onset of symptoms 3 hours prior while taking a shower at home. She had a history of left middle cerebral artery lacuna stroke 5 years ago with few neurologic sequel. She could walk without support and lived independently. She also has the disease of atrial fibrillation and was on a regimen of 2.5 mg of warfarin once daily. However, her prothrombin time was 16.45 sec, international normalized ratio (INR) 1.43, which was under the suggested therapeutic range (INR 2.0e3.0).2 On presentation, she was conscious but agitated. She complained of intolerable pain in her bilateral buttocks and thighs. She also complained of numbness and weakness in her bilateral legs. Her blood pressure (136/88 mm Hg) and other vital signs were normal at triage. A neurologic examination showed that she had intact cranial nerve function and normal muscle power and sensation in both upper limbs. However, there were paraplegia of 1

2 Case Report

both legs, decreased pin-prick sensation, and hyperalgesia below L1 dermatome. She also had decreased bilateral knee jerk reflex. The lumbar spine X-ray revealed normal alignment. Lumbar spine magnetic resonance imaging (MRI) was arranged because there was an initial strong suspicion of lumbar spine myelopathy. The patient maintained complaining of intractable pain despite the use of analgesics while waiting for her MRI. Comprehensive physical evaluation showed diminished bilateral femoral artery pulsation and decreased bilateral distal pulsation. Her feet were cold and pale. With aforementioned finding, acute bilateral legs ischemia due to an abdominal aortic lesion, such as aortic dissection or aortic occlusive disease, was suspected. An abdominal computed tomography with contrast revealed a filling defect of the low abdominal aorta just above the aortic bifurcation of the common iliac artery (Fig. 1, white arrows). There were also occlusive lesions over the bilateral femoral arteries (Fig. 2, white arrows). A diagnosis of acute bilateral ischemia in the legs due to occlusion of the aortoiliac artery and bilateral femoral arteries was made about 3 hours after she came to our ED. Emergent surgery with percutaneous endovascular thrombectomy of the abdominal aorta, bilateral femoral arteries, and distal arteries of the lower legs was performed 1.5 hours after the diagnosis was made. Her pain was rapidly relieved after surgery, and she recovered her muscle power fully. Two-dimensional transthoracic echocardiography was performed which revealed mild mitral regurgitation without patent foramen ovale. She was discharged from our hospital 6 days later with a prescription for oral warfarin and was symptom free when she was followed up after 6 months.

Annals of Vascular Surgery

Fig. 1. The reconstructed computed tomography image reveals filling defect at low abdominal aorta just above aortic bifurcation of common iliac artery (white arrow).

DISCUSSION Aortoiliac occlusive disorder, often related to an atherosclerotic disease, usually presents as chronic intermittent claudication and pain in the buttocks. The occlusion develops gradually, resulting in the dilation of several collateral vessels. Distal flow circulation may therefore be preserved even with critical stenosis of the low abdominal aorta. Increased oxygen demand during exercise in combination with a limited blood supply can also explain the worsening of claudication and pain during exercise.3 In this case, the patient developed paraplegia and pain within minutes without chronic claudication or buttocks pain. This is different from gradually developed atherosclerotic aortoiliac occlusion, as previously reported.1,4,5 Comparing with the case that was described by Schroder et al.6, our patient had no history of severe peripheral artery disease and developed critical limb ischemia more rapidly. Considering her history of ischemic stroke and atrial fibrillation, acute arterial embolic occlusion superimposed on preexisting atherosclerotic disease

Fig. 2. White arrows indicates bilateral femoral artery severe stenosis with poor enhancement by contrast medium.

stenosis was a more reasonable mechanism of sudden aortoiliac occlusion. The initial presentation of acute bilateral leg weakness and paresthesia misled us toward a diagnosis of spinal cord disease, such as spinal infarction, acute transverse myelitis, or a different spinal myelopathy. It was previously reported that such patients were more frequently referred to neurologists rather than to vascular surgeons.7 Previous case series also reported a mean delay of 24 hours from the time of presentation until diagnosis.8

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Table I. Comparison of acute spinal myelopathy and acute aortoiliac occlusion Acute spinal myelopathy

Onset Pain

Acute or sudden Back, lumbar, or thoracic spine Severity of pain Moderate to severe Knee jerk Increased Muscle power Decreased Sensation Abnormal Distal pulsation Normal Skin color Normal Temperature Warm

Acute aortoiliac occlusion

Sudden Buttocks and thighs Intolerable Decreased Decreased Abnormal Decreased Pallor or cyanosis Cold

made it possible to achieve an outstanding functional recovery. In conclusion, distinguishing aortic lesions from myelopathy in paraplegia is difficult. Early recognition of these patients is possible with careful physical examination focused on distal pulsation and deep tendon reflex and abnormal presentation of unusual pain. Patients with risk factors of arterial embolism, such as atrial fibrillation and valvular heart disease, are more likely to present with symptoms of acute arterial occlusion. We report this case to remind physicians to always consider aortic disorder during differential diagnosis of acute paraplegia. REFERENCES

However, the intractable pain in the limbs of our patient could not readily be explained by acute spinal myelopathy. With careful physical examination, we found symptoms of limb ischemia such as diminished pulsation, cold, and pallor in the limbs. Although different degrees of limb weakness and paresthesia are observed in spinal myelopathy and in limb ischemia, neurologic presentation of acute limb ischemia can be distinguished from acute myelopathy by upper motor neuron signs which manifest as increased deep tendon reflex in myelopathy and decreased reflex in ischemic legs, as seen in our patient. Table I displays the difference of acute spinal myelopathy and acute aortoiliac occlusion. Delay in diagnosis and treatment of acute limb ischemia leads to poor prognosis, higher mortality, lifelong paraplegia, and amputation.8,9 Careful physical evaluation of this case enabled early recognition of a vascular emergency rather than spinal myelopathy. We took only 3 hours to arrive at the diagnosis and 5 hours for revascularization, which

1. Leriche R, Morel A. The syndrome of thrombotic obliteration of the aortic bifurcation. Ann Surg 1948;127:193e206. 2. Wann LS, et al. 2011 ACCF/AHA/HRS focused update on the management of patients with atrial fibrillation (update on Dabigatran): a report of the American College of Cardiology Foundation/American Heart Association Task Force on practice guidelines. Circulation 2011;123:1144e50. 3. Muller MD, Reed AB, Leuenberger UA, Sinoway LI. Physiology in medicine: peripheral arterial disease. J Appl Physiol 2013;115:1219e26. 4. Wang YC, Chiu YS, Yeh CH. Leriche’s syndrome presenting as sciatica. Ann Vasc Surg 2010;24:694.e1e3. 5. Lipetz JS, Beer JR, Silber JS. Atypical proximal limb pain of suspected high lumbar stenotic origin arising from severe aortoiliac diseasedLeriche’s syndrome. Pain Physician 2004;7: 123e8. 6. Schroder M, et al. Acute painless paraplegia of the legs as a manifestation of extensive acute Leriche syndrome. Clin Res Cardiol 2007;96:240e2. 7. Cowan KN, Lawlor DK. Sudden onset of paraplegia from acute aortic occlusion: a review of 2 cases and their unique presentation. Am J Emerg Med 2006;24:479e81. 8. Meagher AP, et al. Acute aortic occlusion presenting with lower limb paralysis. J Cardiovasc Surg (Torino) 1991;32: 643e7. 9. Akhaddar A, et al. Acute paraplegia revealing Leriche syndrome. Intern Med 2012;51:981e2.

Aortoiliac occlusive disease presenting as sudden onset paraplegia.

Thromboembolism and atherosclerotic stenosis both can cause arterial occlusion. Aortoiliac occlusive disease involving bifurcation of the aortoiliac a...
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