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J Thorac Cardiovasc Surg 2001;121:998-1001
© 2001 The American Association for Thoracic Surgery

Brief Communications

The implications of extensive cerebral vascular dysplasia in surgical repair of coarctation of the aorta and ventricular septal defect

From the Cardiothoracic Unit,^a Neurosciences Unit,^b and Radiology and Physics Units,^c Institute of Child Health (University College London) and Great Ormond Street Hospital for Children NHS Trust, London, United Kingdom.

Dr Hsia is the 1999 National Science Foundation International Postdoctoral Research Fellow (INT-9802808). MR research is supported by the Wellcome Trust (035352).

Received for publication July 21, 2000. Accepted for publication July 28, 2000. Address for reprints: Victor Tsang, FRCS, Cardiothoracic Unit, Great Ormond Street Hospital for Children, Great Ormond St, London WC1N 3JH, United Kingdom (E-mail: victor.tsang@gosh-tr.nthames.nhs.uk).

Although acute neurologic problems are relatively common postoperatively in patients with arch anomalies, ¹ associated anomalies of the head and neck vessels are not routinely excluded preoperatively. In this report we describe the case of a patient with severe dysplasia of the intracerebral vessels and incomplete circle of Willis who underwent successful concomitant repair of a long-segment aortic coarctation and ventricular septal defect (VSD) under hypothermic circulatory arrest. Despite a satisfactory cardiovascular outcome, he had a cerebral infarction during follow-up. Modern magnetic resonance (MR) methods may assist in the diagnostic work-up of patients with arch anomalies, including coarctation. Ischemic stroke caused by cerebroarterial disease in children with congenital heart disease will also be discussed.

Clinical summary

An asymptomatic, term, 9-month-old boy weighing 11 kg was referred for evaluation of a grade 3/6 pansystolic heart murmur. Transthoracic echocardiography demonstrated normal situs and intracardiac connections. A perimembranous VSD, partially plugged by accessory tricuspid valvular tissue and with 4 m/s left-to-right flow, was seen. However, the left-sided aortic arch was tortuous, with increased flow velocity up to 4 m/s near the origin of the left common carotid artery, suggesting the presence of a coarctation. Cardiac catheterization confirmed the VSD and demonstrated an aortic coarctation with a prominent ductal dimple at the site of the usual origin of the left common carotid artery, which was absent. Instead, its perfusion territory was supplied by multiple small collaterals. Furthermore, the right subclavian artery had an aberrant origin from an unusually low position on the descending aorta. Because of these irregularities, MR angiography was performed, demonstrating markedly tortuous, dilated, and dysplastic proximal intracranial . . . [Full Text of this Article]

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