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      Takayasu arteritis presenting with massive cerebral ischemic infarction in a 35-year-old woman: a case report

      case-report
      1 , 1 ,
      Journal of Medical Case Reports
      BioMed Central

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          Abstract

          Introduction

          Takayasu arteritis is a relatively rare type of large-vessel arteritis that primarily affects the aorta and its major branches, the coronary arteries, and the pulmonary arteries. Depending on the different groups of blood vessels involved in the disease process, the clinical presentation of Takayasu arteritis varies. Here we report a case of a woman presenting with a debilitating massive cerebral ischemic infarct that turned out to be a relatively rare first presentation of Takayasu arteritis.

          Case presentation

          A 35-year-old Chinese woman presented to the Emergency Department with left hemiparesis, pain and numbness of her arms and weak radial pulses. Her laboratory results showed an elevated C-reactive protein and erythrocyte sedimentation rate, and subsequent digital subtraction angiography demonstrated narrowing and occlusion of the major branches of her aortic arch. We report the case of a patient with Takayasu arteritis presenting with a massive cerebral ischemic infarct and review the current literature on this topic.

          Conclusion

          Takayasu arteritis is a relatively rare disease with various and sometimes devastating clinical manifestations, such as massive cerebral ischemic infarction as in our case. Currently, there are multiple diagnostic tools and treatment options available, and more under investigation. Early, appropriate diagnosis and initiation of proper therapy could avoid further progression and reduce complications of the disease.

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          Most cited references24

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          Takayasu arteritis: a review.

          Takayasu arteritis is a well known yet rare form of large vessel vasculitis. This review details the history, clinical features, differential diagnoses, classification, and immunology of the disorder. Suppression of inflammation and preservation of vascular competence are the aims of treatment. As with any rare disease, randomised controlled treatment trials are either lacking or based on small patient numbers, making management decisions difficult. Current evidence based treatments are presented and discussed.
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            Diagnostic criteria for Takayasu arteritis.

            Diagnosis of Takayasu arteritis (TA) is often delayed because of a non-specific clinical presentation. Ishikawa's criteria (1988) has been widely used for the diagnosis of this disease. Few modifications have been proposed in Ishikawa's criteria for the diagnosis of TA. The proposed modifications include: (a) removal of the obligatory criteria of age less than 40 years; (b) inclusion of characteristic signs and symptoms as a major criteria; (c) removal of age in defining hypertension; (d) deletion of the absence of aorto-iliac lesion, in defining abdominal aortic lesion; and (e) an addition of coronary artery lesion in absence of risk factors. The criteria proposed consists of three major criteria including left and right mid subclavian artery lesions and characteristic signs and symptoms of at least one month duration and ten minor criteria-a high erythrocyte sedimentation rate, carotid artery tenderness, hypertension, aortic regurgitation or annuloaortic ectasia, pulmonary artery lesion, left mid common carotid lesion, distal brachiocephalic trunk lesion, descending thoracic aorta lesion, abdominal aorta lesion and coronary artery lesion. Presence of two major or one major and two minor criteria or four minor criteria suggests a high probability of TA. When applied to 106 Indian patients of angiographically proven TA and 20 control subjects, it had a sensitivity of 92.5% and specificity of 95% that was higher than that of Ishikawa's criteria (sensitivity 60.4%, specificity 95%) and American college of Rheumatology criteria (sensitivity 77.4%, specificity 95%). Similarly, this criteria had a 96% sensitivity and 96% specificity in 79 Japanese patients of TA and 79 control subjects. Adoption of these criteria is expected to prevent the possibility of an under diagnosis of TA.
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              Early diagnosis and follow-up of aortitis with [(18)F]FDG PET and MRI.

              The aim of this prospective study was to compare fluorine-18 fluorodeoxyglucose ([(18)F]FDG) positron emission tomography (PET) with magnetic resonance imaging (MRI) in patients with early aortitis, at the time of initial diagnosis and during immunosuppressive therapy. The study population consisted of 15 patients (nine females and six males; median age 62 years, range 26-76 years) who presented with fever of unknown origin or an elevated erythrocyte sedimentation rate or elevated C-reactive protein and who showed pathological aortic [(18)F]FDG uptake. Fourteen of these patients had features of early giant cell arteritis (GCA), while one had features of early Takayasu arteritis. During follow-up, seven PET scans were performed in six patients with GCA 4-30 months (median 19 months) after starting immunosuppressive medication. The results of [(18)F]FDG imaging were compared with the results of MRI at initial evaluation and during follow-up and with the clinical findings. At baseline, abnormal [(18)F]FDG uptake was present in 59/104 (56%) of the vascular regions studied in 15 patients. Seven follow-up PET studies were performed in six patients. Of 30 regions with initial pathological uptake in these patients, 24 (80%) showed normalisation of uptake during follow-up. Normalisation of [(18)F]FDG uptake correlated with clinical improvement and with normalisation of the laboratory findings. All except one of the patients with positive aortic [(18)F]FDG uptake were investigated with MRI and MRA. Thirteen of these 14 patients showed inflammation in at least one vascular region. Of 76 vascular regions studied, 41 (53%) showed vasculitis on MRI. Of 76 vascular regions studied with both PET and MRI, 47 were concordantly positive or negative on both modalities, 11 were positive on MRI only and 18 were positive on PET only. MRI was performed during follow-up in six patients: of 17 regions with inflammatory changes, 15 regions remained unchanged and two showed improvement. Whole-body [(18)F]FDG PET is valuable in the primary diagnosis of early aortitis. The results of [(18)F]FDG PET and MRI in the diagnosis of aortitis in this study were comparable, but FDG imaging identified more vascular regions involved in the inflammatory process than did MRI. In a limited number of patients [(18)F]FDG PET was more reliable than MRI in monitoring disease activity during immunosuppressive therapy.
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                Author and article information

                Contributors
                Journal
                J Med Case Rep
                J Med Case Rep
                Journal of Medical Case Reports
                BioMed Central
                1752-1947
                2013
                5 July 2013
                : 7
                : 179
                Affiliations
                [1 ]Intensive Care Unit, Shanghai First People's Hospital, School of Medicine, Shanghai Jiao Tong University, 650 Xinsongjiang Road, Shanghai 201620, China
                Article
                1752-1947-7-179
                10.1186/1752-1947-7-179
                3707746
                23830048
                df2a50b9-383f-43c3-b353-0add133d5bc6
                Copyright ©2013 Gao and Wang; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 5 March 2013
                : 1 May 2013
                Categories
                Case Report

                Medicine
                Medicine

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