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• W2087702905 abstract "We were very interested to read the article by Ghez and colleagues [1Ghez O.Y. Chetaille P.M. Campbell B.J. Van Praagh R. Metras D. Tetralogy of Fallot with aortic valvular stenosis surgical correction in one case.Ann Thorac Surg. 2002; 73: 967-969Abstract Full Text Full Text PDF PubMed Scopus (10) Google Scholar]. We would like to add another case to the four cases cited in the literature. In 1995, a 38-week-gestation male was postnatally diagnosed with pulmonary atresia, ventricular septal defect, and critical aortic stenosis. Echocardiography had shown severe aortic valve stenosis with a doming bicuspid aortic valve and a Doppler gradient in excess of 80 mm Hg. There was biventricular hypertrophy with a thick interventricular septum. The ventricular septal defect was in the perimembranous area and there was overriding of the aorta. The small-sized pulmonary arteries were continuous and fed by a patent ductus arteriosus from a left aortic arch. Cardiac catheterization confirmed the anatomy and significant valvar aortic stenosis with a 60 mm gradient. No major aortopulmonary collaterals were identified. He also had an elevated conjugated bilirubin and elevated liver enzymes. A HIDA scan had not indicated biliary atresia. His subsequent course has been consistent with a diagnosis of Alagille syndrome. Hypocalcemia later developed, and he had evidence of hypoparathyroidism, although chromosomal analysis was normal with no evidence of 22q deletion. At 18 days of age, this 2.9-kg baby underwent an aortic valvotomy and placement of a 3.5-mm right ventricle to pulmonary artery conduit with ligation of the patent ductus arteriosus. The diameters of pulmonary arteries were noted to be less than the diameter of the graft. Under circulatory arrest, the trileaflet aortic valve was visualized. A commissurotomy was performed. At 4 months of age, the patient underwent a 10-mm right ventricle to pulmonary artery conduit replacement with enlargement of the proximal left pulmonary artery. The right and left pulmonary arteries were approximately 6 mm in diameter. He underwent bilateral balloon pulmonary angioplasties at 7 months. There was a gradient of approximately 35 to 40 across a thickened aortic valve. The proximal right pulmonary artery required redilation at 13 months and again at 1 year later. During this time, there was only a 15-mm peak to peak gradient across the aortic valve with trivial aortic valve insufficiency. At 23 months he underwent enlargement of his ventricular septal defect with tunneling of this into the aorta, creation of an atrial septal defect to act as a fenestration, and another right ventricle to pulmonary artery conduit. A 16-mm aortic valve homograft with a 16-mm distal Goretex tube extension was used. At 4.5 years of age at catheterization, the boy's right ventricular pressures were elevated to 70 mm Hg systolic compared with the left ventricular pressures of 90. There was mild stenosis at the bifurcation of the left and in the right main pulmonary artery. The functionally bicuspid aortic valve had mild insufficiency and minimal stenosis. At 8 years of age, he had further dilation of the left pulmonary artery, and a stent was placed in the proximal right artery. He remains with elevated right-sided pressures but is asymptomatic. He may require aortic valve and conduit replacement in the future. Aortic valvotomy and a right ventricle to pulmonary artery conduit was successful in our case [2Martin R.P. Radley-Smith R. Yacoub M.H. Pulmonary atresia and aortic valve stenosis.Int J Cardiol. 1987; 16: 103-105Abstract Full Text PDF PubMed Scopus (5) Google Scholar, 3Patel R.G. Freedom R.M. Bloom K.R. Rowe R.D. Truncal or aortic valve stenosis in functionally single arterial trunk. A clinical, hemodynamic, and pathologic study of six cases.Am J Cardiol. 1978; 42: 800-809Abstract Full Text PDF PubMed Scopus (24) Google Scholar]. There was no main pulmonary artery and with the hypoplastic right and left pulmonary arteries, a systemic artery to pulmonary artery shunt could have resulted in severe stenosis or occlusion of the branch pulmonary arteries. A systemic artery to pulmonary artery shunt may also have resulted in poorer diastolic perfusion pressures to an already hypertrophied myocardium. We have not had the development of subvalvular left ventricular hypertrophy needing surgery, as was described by Ghez and colleagues, possibly because of the aortic valvotomy done in the neonatal period. We would like to add our experience regarding this unusual association to those of Ghez and colleagues. ReplyThe Annals of Thoracic SurgeryVol. 78Issue 5Preview Full-Text PDF" @default.
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• W2087702905 date "2004-11-01" @default.
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• W2087702905 title "Successful Repair of Tetralogy of Fallot With Aortic Valvular Stenosis" @default.
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• W2087702905 doi "https://doi.org/10.1016/j.athoracsur.2003.10.131" @default.
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