Takayasu arteritis of subclavian artery in a Caucasian
Ramesh Gowda2004, International Journal of Cardiology
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Takayasu arteritis, an inflammatory and obliterative disease of medium and large arteries, is classified as a giant cell arteritis. It has a predilection for the aortic arch, its main branches, and coronary and pulmonary arteries. The early symptoms of Takayasu arteritis may be mainly systemic and may resemble polymyalgia rheumatica. The etiology is unknown but autoimmunity has been suggested to play a role. Diagnosis is based on symptoms, physical findings, and imaging, because tissue diagnosis is rarely feasible. Unlike atherosclerotic vascular disease, Takayasu arteritis affects primarily, but not exclusively, young women. Contrary to earlier reports, it is not limited to the women of Japanese origin but is present worldwide. The current report is of a Caucasian woman who presented with nonspecific complaints of upper back pain, weakness, malaise, and fatigue. Her physical examination revealed absent left radial pulse and a blood pressure differential, later confirmed by radiological imaging studies to be due to left subclavian artery stenosis consistent with Takayasu arteritis. The presentation and management of the patient is described, and Takayasu arteritis is succinctly reviewed.
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Introduction
Takayasu arteritis, also known as pulseless disease, is an unusual cause of symptomatic peripheral vascular disease. It was first reported in 1908 by a Japanese ophthalmologist Takayasu, who described retinal arteriovenous shunts which, appeared in a wreath-like distribution around the optic disc and microaneurysms of the retinal vessels in a 19-year-old Japanese woman [1]. Later, similar findings were found in association with cool, pulseless upper extremities. Since that time the syndrome has been described as Takayasu arteritis. It is a more widespread disease than was previously stated, and although more common in young women of Japanese origin, it is actually present worldwide in all age groups with variegated patterns of clinical and angiographic presentation [2]. However, two predominant forms may be identified: the Japanese form, which involves the aortic arch, and the Indian form, in which vasculitis is present in abdominal aorta and its branches above the renal arteries with an upright extension to the thoracic aorta and a protean clinical picture with a more systemic spectrum. We report a case of Takayasu arteritis of left subclavian artery in a Caucasian young woman who presented with nonspecific systemic symptoms, and briefly discuss the subject.
Case presentation
A 30-year-old Caucasian female presented with upper back pain between shoulder blades. She also had few months' history of weakness, malaise, and fatigue. On questioning, the patient did admit to a feeling of weakness and heaviness of the left arm when she used her arm over the head, for instance during combing her hair. She smoked cigarettes previously. On examination, there was a significant asymmetry in the pulses and blood pressure in the upper extremities. The left radial pulse was absent. The blood pressure in right arm was 120/80 mmHg and in left arm it was 98/68 mmHg. She had a bruit over the left subclavian region. The cardiac, respiratory, abdominal, and neurological examinations were all otherwise unremarkable. The laboratory parameters included a white blood count of 8700 per ul, hemoglobin of 13.1 g/dl, and platelet count of 291,000 per ul. The erythrocyte sedimentation rate was 63 mm/h. The results of antinuclear antibody, rheumatoid factor, antidouble-strand DNA, Anti-SS-A and SS-B, were all negative. The electrocardiogram showed sinus and was unremarkable.
Magnetic resonance imaging of the chest with gadolinium revealed diffusely narrowed, abnormal appearing left subclavian artery ( Fig. 1). On arteriogram of upper extremities, the left subclavian artery and proximal portion of the left axillary artery were diffusely but smoothly narrowed ( Figs. 2 and 3). The left vertebral artery was not opacified, and was considered occluded. The aorta and all the other major arteries were widely patent. Although the aorta seemed normal, the nature of narrowing of involved arteries along with age and sex of the patient appeared to be consistent with Takayasu arteritis. The patient was started on oral corticosteroids, and methotrexate was added subsequently. After 2 months, the repeat erythrocyte sedimentation rate was 5 mm/h. She progressively has shown improvement in symptoms and was doing well at 7-month follow-up.
Figure 1
Magnetic resonance imaging of the chest with gadolinium contrast: T1 coronal image showing diffusely narrowed abnormally appearing left subclavian artery from its origin to the junction with axillary artery. The findings were confirmed by 3D reconstruction.
Figure 2
Aortic arteriogram showing significant narrowing of the left subclavian artery, with normal right innominate and bilateral carotid branches. Left vertebral artery is not visualized.
Discussion
Takayasu arteritis is a chronic vasculitis of unknown etiology classically leading to vascular insufficiency of the upper extremities. It predominantly affects women (80 -90%) between ages 10 and 40 years [3]. Almost all patients have roots from Asian or South American countries, which has led to a search in the role of genetic factors in its pathogenesis [4]. The incidence in US and Europe is of one to three new cases per million per year [3]. Takayasu arteritis involves primarily the aorta, its main branches, and the coronary and pulmonary arteries [5]. The inflammation may be localized to a portion of the vessel or involve the entire vessel. An immunogenetic association has been suggested for this condition [6]. In addition, a positive association of Takayasu arteritis with HLA-B52 and HLA-B39 has been observed [6]. Pathogenesis of Takayasu arteritis is poorly understood, but is thought to be a cell-mediated process. In one study, the infiltrating cells present in Takayasu arteritis effected vessels were consisted of killer cells and especially gamma delta T lymphocytes [7]. In another study, antiendothelial antibodies were suggested to have a role in pathogenesis as they were found in high titers [8]. Besides, an increased in serum soluble E-selectin and thrombomodulin have been reported, but their value in routine diagnosis or follow-up is uncertain [9,10]. Anemia of chronic disease and marker of nonspecific inflammatory process, including an elevated erythrocyte sedimentation rate, increased serum C-reactive protein and alpha-2 globulin, and hypoalbuminemia are often present, but usually there is no increase in the white cell count [8].
Systemic symptoms of fatigue, loss of weight, and lowgrade fever are common in early phase, and vascular symptoms are rare at presentation. Patients often look chronically ill. Narrowing, occlusion, and dilatation of the affected arteries cause a variety of vascular symptoms. Reduced blood pressure in one or both arms is common with a differential of >10 mmHg between the arms. Diminished and asymmetrical arterial pulses and bruits over the affected arteries are usually heard.
Diagnosis of Takayasu arteritis is often challenging because the clinical features are similar to those of other systemic inflammatory diseases. Detection of bruits or decreased pulses, especially in a young woman should narrow the focus of diagnosis. Transthoracic and transesophageal echocardiography are useful in delineating the lesions affecting the aorta [11]. A familiarity with the radiographic, computed tomographic, magnetic resonance imaging, and angiographic features of Takayasu arteritis may permit and earlier diagnosis and treatment [12]. In early-phase of Takayasu arteritis (pre-pulseless phase), computed tomography and magnetic resonance imaging show thickening of the aortic wall. Anatomically, the latephase Takayasu arteritis has been classified into four types: classic pulseless disease (type I), a mixed type (type II), the atypical coarctation type (type III), and the dilated type (type IV) [12]. Arteriography is usually needed to confirm the diagnosis of the late-phase of the disease, where it usually demonstrates luminal changes such as stenosis, occlusion, or aneurysmal dilatation of the involved artery and their branches. Although a clear outline of the involved arterial walls is depicted by arteriography, the arterial wall thickening is not shown, which limits the value of angiography in diagnosing the early phase of the disease when the luminal changes have not taken place. The techniques of computed tomography and magnetic resonance imaging have improved significantly over the time and may eventually replace arteriography, as in one study, the computed tomography was 95% sensitive and 100% specific and was more sensitive than conventional arteriography to detect mural vessel changes in Takayasu arteritis [13]. Besides, computed tomography and magnetic resonance imaging can be conveniently used to during follow-up [14].
An improvement in the clinical findings and subsidence of the active inflammatory process can be expected with an early steroid treatment, which is the mainstay of the therapy for Takayasu arteritis. Corticosteroids are effective in suppressing the systemic symptoms, arresting the progression of disease, and reverting the anemia and elevated acute phase reactants [8]. Various cytotoxic drugs like methotrexate and cyclophosphamide have been used with variable success [15]. Mycophenolate mofetil may be an alternative to steroids and cytotoxic drugs in patients intolerant to these agents [16]. Erythrocyte sedimentation rate is the best marker to follow the course of the disease and its response to medical treatment. Angioplasty or bypass grafting may be needed in late cases when irreversible stenosis occurs with significant fibrosis and ischemic symptoms [2,17].
Conclusion
Takayasu arteritis is a rare disease in Caucasians with a heterogeneous presentation, progression, and response to therapy. Erythrocyte sedimentation rate is the best marker to follow the course of the disease and its response to medical treatment.
Related topics
CardiologyMagnetic Resonance ImagingMedicineBlood PressureBack PainEtiologyPeripheral Vascular DiseaseYoung WomenPhysical examinationEuropean Continental Ancestry GroupPolymyalgia RheumaticaGiant Cell ArteritisArteritisAortic archSubclavian arteryPulmonary ArteryVascular DiseaseCardiovascular medicine and haem...