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CASE REPORT
Year : 2007  |  Volume : 10  |  Issue : 3  |  Page : 178-181
 

Aortoiliac occlusive disease masquerading as cerebrovascular accident


1 Department of Neuropathology, National Institute of Mental Health and Neurosciences, Bangalore, India
2 Department of Neurology, National Institute of Mental Health and Neurosciences, Bangalore, India

Correspondence Address:
S K Shankar
Department of Neuropathology, National Institute of Mental Health and Neurosciences, Bangalore - 560 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-2327.34799

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   Abstract 

Acute aortoiliac occlusion is an unusual but potentially catastrophic condition causing acute limb ischemia and associated with early and high rates of mortality and morbidity. It is caused by either embolic occlusion of the infra renal aorta at the bifurcation or beyond or thrombosis of the abdominal aorta and its large terminal branches. Neurological symptoms are rare manifestation of acute aortoiliac occlusion and when neurological symptoms predominate, patients are mistakenly considered to have cerebrovascular event. We present a 60-year-old man with atherosclerotic thrombotic occlusion of the left common iliac artery causing acute painful monoplegia. We mistook the acute monoplegia due to acute limb ischemia for cerebrovascular accident. Pathologic examination revealed a firm thrombus occluding the origin of left common iliac artery and extending along the length of the vessel. Acute aortic iliac occlusion can masquerade as a cerebrovascular stroke and a thorough clinical evaluation and imaging studies allow early diagnosis and instituting life-saving treatment timely.


Keywords: Acute limb ischemia, aortoiliac occlusive disease, atherosclerosis, Leriche′s syndrome, limb weakness


How to cite this article:
Nandeesh B N, Mahadevan A, Tandon A, Satishchandra P, Shankar S K. Aortoiliac occlusive disease masquerading as cerebrovascular accident. Ann Indian Acad Neurol 2007;10:178-81

How to cite this URL:
Nandeesh B N, Mahadevan A, Tandon A, Satishchandra P, Shankar S K. Aortoiliac occlusive disease masquerading as cerebrovascular accident. Ann Indian Acad Neurol [serial online] 2007 [cited 2020 Mar 28];10:178-81. Available from: http://www.annalsofian.org/text.asp?2007/10/3/178/34799



   Introduction Top


Acute aortoiliac occlusion is an infrequent but potentially catastrophic condition with an early and a high mortality rate. It is caused by either embolic occlusion of the infra renal aorta at the bifurcation or beyond or thrombosis of the abdominal aorta and its large branches. We report a patient who died 18h after presenting with left lower limb weakness and at autopsy, it was found to be due to acute iliac obstruction secondary to a thrombosis developing in the left common iliac artery in the setting of severe atherosclerosis.


   Case Report Top


A 60-year-old man presented to the neurological emergency services with sudden onset of weakness of left lower limb of 12h duration, which was preceded by pain. The weakness gradually progressed with persistence of pain, localized to the left side of lower back radiating to the mid portion of thigh. There was no history of trauma, head injury, bladder symptoms or seizures. Patient was not previously detected to be a diabetic or a hypertensive.

On general physical examination, patient was moderately built and nourished, afebrile with BP 160/100 mm Hg, respiratory rate 16/min, radial pulse rate 80/min, regular, normal volume, while feeble pulsations were felt in the left lower limb. Cardiovascular and respiratory system examination was non-contributory. There was no organomegaly or palpable masses felt in the abdomen. Examination of the skeletal system including the neck and spine were normal. Movement of the left hip and thigh elicited local tenderness. Neurological examination revealed a conscious, alert patient responding to verbal commands. Ocular movements were normal. On examination of the motor system, muscle power was decreased in the left lower limb (0-1/5, MRC grade) and normal in the other limbs. Sensory system examination was essentially normal. The patient kept the left hip extended and abducted in resting position. Superficial and deep tendon reflexes were elicitable normally while the plantar response was mute on the left side. There were no meningeal signs or pyramidal signs. Acute onset of monoplegia in an elderly male prompted a clinical diagnosis of cerebrovascular accident in the territory of anterior cerebral artery. Cranial computerized tomography scan was normal except for mild dilatation of the ventricles. X-ray of the pelvis was normal. Biochemical parameters, except for mild elevation of SGOT, SGPT and blood urea levels, were normal. The Creatine phosphokinase was elevated (22,700U/L). Before further investigations could be carried out, the patient succumbed to cardiorespiratory arrest 18h after the onset of symptoms.

Complete autopsy confined to examination of internal viscera and brain (excluding spinal cord) was performed four hours post mortem following informed consent from close relatives. External examination of the body did not reveal any positive findings. The heart weighed 350 gms with gross atherosclerosis of the aorta and hypertrophy of the left ventricle. The left main coronary artery was eccentrically narrowed by atherosclerosis without thrombotic occlusion. The abdominal aorta and the iliac arteries had extensive atherosclerosis with calcification and focal ulceration. A firm thrombus occluding the origin of left common iliac artery was noted, extending along the length of the vessel [Figure - 1]A, B sparing the right common iliac artery. Histology of the iliac arteries revealed atheromatous plaques [Figure - 2]A, B with ulceration, calcification and a large fresh thrombus adherent to the sub-endothelial zone [Figure - 2]A. The adventitia had mixed chronic inflammatory cell infiltrates of moderate density. The arch of the aorta, mesenteric and renal arteries had atherosclerosis with calcification, but the lumen was wide and patent. Pulmonary edema and marked congestion of kidneys, spleen, GIT, liver and adrenals was noted reflecting terminal congestive cardiac failure. Benign nephrosclerotic changes secondary to hypertension was observed in the kidneys.

The brain was edematous and had focal ischemic lesions. The vessels of circle of Willis had atherosclerosis all along the length, but no thrombotic occlusion. The upper cervical cord was essentially normal. Histology and histochemical study of ilio-psoas muscle revealed subsarcolemmal accumulation of mitochondrial enzyme reaction product and variation in fiber size. Modified Gomori's trichrome stain highlighted ragged red granular deposits in subsarcolemmal zone in many fibers. These features in the skeletal muscle close to the iliac occlusion reflect hypoxic mitochondrial cytopathy as a secondary phenomenon to vaso-occlusive pathology.


   Discussion Top


Acute lower limb ischemia continues to be a diagnostic challenge in emergency services. Aortoiliac or iliac artery occlusive disease (AIOD) condition is an important and devastating cause of acute lower limb ischemia with high rate of mortality and morbidity, requiring prompt surgical intervention like thrombectomy / embolectomy. Aortoiliac or iliac artery occlusive disease forms a part of the vast group of peripheral arterial diseases, having a prevalence of 3-30%, majority having multiple risk factors.

Aortoiliac occlusive disease occurs when aorta / iliac arteries become narrowed or blocked by embolic occlusion of the infra-renal aorta at the bifurcation or beyond or acute thrombosis of the abdominal aorta and its terminal branches. Usually patients with AIOD have multiple concomitant pathologies of heart, lungs and diabetes mellitus. Primary thrombosis accounts for majority of cases of acute aortic occlusion. Seventy five to eighty percent of thrombotic aortic occlusion occur in the setting of underlying severe atherosclerotic aortoiliac occlusive disease as seen in this case and are frequently precipitated by a low flow state secondary to heart failure or dehydration. Atherosclerotic plaques may induce symptoms either by obstructing blood flow or by breaking apart and embolizing the atherosclerotic and/or thrombotic debris to more distal blood vessels. The composition of the atherosclerotic plaque, rather than the degree of stenosis, appears to be a critical predictor for both the risk of plaque rupture and subsequent thrombogenicity. [1] Plaque rupture can also be precipitated by abdominal trauma leading onto thrombosis and occlusion. [2]

Thrombosis unrelated to atherosclerotic disease can occur at foci of vessel injury, as with invasive catheters, balloons, sites of bypass grafting or intra-arterial drug injection and in the setting of aneurysm. Takayasu's disease, hypercoagulable states are rarer causes of AIOD. [3] In more than 85% of symptomatic patients, the zone of involvement of aortoiliac occlusion is more dispersed, whereas it is localized in less than 15% of instances, as in the present case. Depending on the level and grade of extension of occlusion of main arteries the collateral circulation may increase, but acute events cause greater damage, as establishment of collaterals take time. Several risk factors exist for development of the arterial lesions that include chronic smoking, high cholesterol level, elevated blood pressure, obesity and family history of heart disease. [5] The recognition of these factors enables physicians to prescribe preventive treatment.

To the best of our knowledge, only one case of thrombotic occlusion of the abdominal aorta (Leriche's syndrome) has been described in the Indian literature. [6] Leriche's syndrome, also referred to as aortic bifurcation occlusion syndrome is due to thrombotic occlusion of abdominal aorta just above the site of its bifurcation (saddle thrombus) and is characterized by inability to maintain penile erection, weakness in both the lower limbs, ischemic pain of intermittent bilateral claudication; absent or diminished femoral pulses, pallor and coldness of both the feet and legs. The present case deviates from the usual Leriche's syndrome by its acuteness, unilaterality of lesion and absence of history of impotence in the male. The tenderness elicited on movement of the left hip and thigh could be due to ischemic spasm of the ilio-psoas and surrounding muscles following sudden occlusion. Muscle histochemistry performed on left ilio-psoas muscle tissue reflects this phenomenon.

Acute occlusion in most cases is heralded by sudden onset of excruciating unilateral / bilateral lower extremity pain that radiates from the mid-thigh distally associated with weakness, numbness and paresthesias. Less common manifestations include sudden onset of lower extremity weakness either unilateral or bilateral, severe hypertension due to renal artery involvement and abdominal pain from mesenteric ischemia. When neurological symptoms predominate patients are often mistakenly thought to have spinal cord infarction or CNS event and the ischemic symptoms may be initially overlooked as happened in the present case. In fact as many as 11-17% of such patients, first undergo neurological or neurosurgical evaluation before the vascular cause is recognized. Most cases on examination reveal cold, pale extremity that are cyanotic and often exhibit a mottled, reticulated and reddish blue appearance that may progress to the blue-black color of gangrene. Pulses are notably absent below the involved segment of the vessel and capillary refill is absent. [4] Persistent ischemia may lead to myonecrosis with elevated creatine phosphokinase levels, secondary hyperkalemia, myoglobinuria and renal acute tubular necrosis. If arterial perfusion is not established within hours, death may supervene due to cardiovascular complications, which is the most important cause of morbidity in these patients with or without treatment. [7]

An arteriogram can help confirm the diagnosis, define the vascular anatomy and degree of perfusion and guide management. Surgical treatment of AIOD is considered an appropriate method of management with favorable outcome. The goals of therapy include restoration of blood flow, preservation of limb and life and prevention of recurrent thrombosis or embolism. [4] Pre-clinical studies have shown that angiogenetic growth factors promote development of collateral arteries referred to as 'therapeutic angiogenesis' and infusion of endothelial progenitor cells, implantation of erythroid progenitor cells - erythroid colony forming cells has been shown to induce angiogenesis and increase blood flow. [8]

The present case with prominent neurological manifestations has made the clinicians overlook the symptoms of tenderness in the thigh and feeble arterial pulsations in the lower limb. An acute presentation and short interval between the onset of symptoms and death has further limited the chances of imaging studies and timely therapeutic intervention.


   Acknowledgments Top


The authors would like to acknowledge Ms. Rajasakti and Mr. Shivaraju for technical assistance. We also thank Mr. Manjunath K for assistance with microphotography.

 
   References Top

1.Corti R, Hutter R, Badimon JJ, Fuster V. Evolving concepts in the triad of atherosclerosis, inflammation and thrombosis. J Thromb Thrombolysis 2004;17:35-44.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Tsai FC, Wang CC, Fang JF, Lin PJ, Kao CL, Hsieh HC. Isolated common iliac artery occlusion secondary to atherosclerotic plaque rupture from blunt abdominal trauma: Case report and review of the literature. J Trauma 1997;42:133-6.  Back to cited text no. 2    
3.Alfrayate JM, Acin F, Bueno A, March JR, Lopez-Quintana A, Cancer S, et al . Aortoiliac thrombosis in antiphospholipid syndrome: Case report. Vasc Endovascular Surg 2002;36:311-5.   Back to cited text no. 3    
4.Tintinall JE, Kelen GD, Stapczynski JS. Emergency medicine. 6 th ed. McGraw Hill: New York; 2004. p. 415-8.   Back to cited text no. 4    
5.Zipes DP, Libby P, Bonow RO, Braunwald E. "Braunwald's heart disease" - A textbook of cardiovascular medicine. 7 th ed. Elsevier Saunders: Philadelphia; 2005. p. 1431-2.  Back to cited text no. 5    
6.Bidwai PS. Thrombotic occlusion of the abdominal aorta (Leriche's syndrome): A case report. J Assoc Physicians India 1961;9:847-52.  Back to cited text no. 6    
7.Surowiec SM, Isiklar H, Sreeram S, Weiss VJ, Lumsden AB. Acute occlusion of the abdominal aorta. Am J Surg 1998;176:193-7.  Back to cited text no. 7    
8.Tateishi-Yuyama E, Matsubara H, Murohara T, Ikeda U, Shintani S, Masaki H, et al . Therapeutic angiogenesis for patients with limb ischemia by autologous transplantation of bone-marrow cells: A pilot study and a randomized controlled trial. Lancet 2002;360:427-35.  Back to cited text no. 8    


    Figures

  [Figure - 1], [Figure - 2]



 

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    Abstract
    Introduction
    Case Report
    Discussion
    Acknowledgments
    References
    Article Figures

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