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Ann Thorac Surg 1999;68:248-250
© 1999 The Society of Thoracic Surgeons
a Clinic for Cardiovascular Surgery, University Cantonal Hospital of Geneva, Geneva, Switzerland
b Clinic for Pediatric Cardiology, University Cantonal Hospital of Geneva, Geneva, Switzerland
Address reprint requests to Dr Kalangos, Clinic for Cardiovascular Surgery, University Cantonal Hospital of Geneva, 24, rue Micheli-du-Crest, 1211 Geneva 14, Switzerland
e-mail: afksendyios.kalangos{at}hcuge.ch
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A 10-year-old Senegalese boy was referred to our institution for investigation of aortic insufficiency. On arrival, he presented with a history of congestive heart failure, New York Heart Association Class III. Blood pressure was 120/50 mm Hg on his left arm and 150/60 mm Hg on his right arm. There was a grade 4/6 diastolic regurgitant murmur, most audible in the mesocardiac area. Chest roentgenogram showed enlarged central opacity, with a cardiac ratio of 60%. Blood analyses were normal, except for an elevated erythrocyte sedimentation rate of 78/h. Transthoracic echocardiography showed marked dilatation of the ascending aorta and severe aortic insufficiency with a markedly dilated annulus of 25 mm. Aortic leaflets were thickened, lacking coaptation during diastole, hence producing a severe central leak. Subsequent thoracic magnetic resonance imaging (MRI) revealed a pear-shaped enlargement of the aortic root (5.15 cm in diameter) and the ascending aorta (Fig 1A). Surgery was conducted using cardiopulmonary bypass (CPB) via the right femoral artery and right atrium, under moderate hypothermia (28°C). The ascending aorta had a whitish appearance, was rigid on palpation, and showed a pear-shaped dilatation with only a slightly dilated aortic caliber just before the origin of the innominate artery. After aortic cross-clamping, cold potassium cardioplegia was directly infused through both dilated coronary ostia. The aortic wall was fibrotic and 3 mm thick. After resection of the dilated ascending aorta, analysis of the aortic valve revealed a dilated aortic annulus and thickened aortic leaflets at their insertion on the annulus. Although the free edges of the leaflets were thin and mobile, preservation of the aortic valve was deemed inappropriate. A Bentall operation was then performed using an aortic cryopreserved homograft 6 cm in length and 23 mm in diameter on the valvular side. A homograft was chosen as the valvular conduit, since this avoids long-term anticoagulation in children who are noncompliant due to problems related to the follow-up in their country of origin. Weaning from CPB was uneventful, and the postoperative course was normal. Histopathological examination of the aortic wall revealed that all three layers were involved in the inflammatory process with disrupted elastic fibers in the media, marked fibrous thickening of the adventitia with obstruction of vaso vasorum, and fibrosis and collagenization of the intima. The media showed some infiltration of mononuclear and giant cells (Fig 1B). Aortic leaflets showed fibrous thickening, inflammation, and patchy vascularized scars on the annular side. Serologic tests for syphilis and tuberculosis were negative, and there were no positive cultures from the aneurysm. These histopathological findings in favor of Takayasu’s arteritis incited us to do a complete investigation of the entire aorta and its major branches. Panaortography revealed stenosis of the left subclavian artery, the right and left common carotid arteries, as well as stenosis of the descending aorta with a moderate poststenotic dilatation (Figs 2A, 2B). The abdominal aorta and its branches were normal.
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ascending aorta is rare and, as in our patient, it is usually associated with disease elsewhere [1]. Mild aortic insufficiency is not uncommon in Takayasu’s arteritis; its incidence varies between 7% and 16% [2]. However, severe aortic insufficiency requiring surgery is rarely reported and is attributed to annular dilatation created by the associated aortic root distension. Moreover, as in our case, nonspecific inflammatory involvement of the aortic leaflets can also contribute to aortic insufficiency by producing leaflet retraction. Aortic dilatation is due to the destruction of elastic fibers of the media, a peculiar finding in Takayasu’s arteritis, or sometimes to the presence of systemic hypertension that weakens the aortic wall with each systole. In cases of aneurysmal formation, high erythrocyte sedimentation rate can be considered as an indicator of ongoing inflammation and destruction of the arterial wall [1]. Aneurysmal lesions are usually found in older patients who have a longer history of the disease.
An aneurysm of the ascending aorta associated with aortic insufficiency as the sole clinical manifestation of Takayasu’s arteritis in a child, as in our case, has been previously described in a 15-year-old girl [4]. We believe that children in whom such a clinical condition is echocardiographically detected merit more detailed evaluation of the entire aorta with its major branches by one of the following imaging methods: computed tomography, MRI, or panaortography. Our case shows that this attitude may not only serve to discover asymptomatic steno-occlusive or aneurysmal lesions, but also contribute to the correct diagnosis by defining the location, extent, and configuration of the lesions. In addition, these radiological findings can affect the therapeutic approach of the patient. Aneurysmal aortitis in Takayasu’s disease of children must be distinguished from other forms of aortitis associated with aneurysm formation. Syphilis usually occurs in adults with positive serologies. The major branches of the aorta are rarely involved, except for an eventual dilatation of the ascending aorta, the arch, or the descending aorta. Giant cell arteritis usually affects older persons and is a widespread arterial disease, commonly involving small- and medium-sized arteries and uncommonly involving the aorta. However, some cases of giant cell arteritis exhibiting pathological features such as arteritis and thrombosis of carotid arteries [4], which are common in Takayasu’s disease, and the fact that giants cells have been observed in some cases of Takayasu’s disease [5], where there is excessive destruction of elastic tissue or fresh thrombus formation, may occasionally create the confusing problem of etiology in many cases of aortitis, thereby forcing pathologists to draw a relationship between these two diseases. Tuberculosis of the aorta commonly causes false or true aneurysms by erosion of the wall, which completely differs from Takayasu’s arteritis in clinical, anatomical, and histological respects.
In conclusion, Takayasu’s arteritis should be considered in the differential diagnosis of ascending aortic aneurysm associated with aortic insufficiency in children. A more complete evaluation of the entire aorta and its major branches allows confirmation of the histopathological suspicion by revealing associated steno-occlusive lesions specific to Takayasu’s disease[3].
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