Cases reported "Tricuspid Atresia"

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1/13. Absent pulmonary valve with tricuspid atresia or severe tricuspid stenosis: report of three cases and review of the literature.

    Absence of the pulmonary valve occurs usually in association with tetralogy of fallot and occasionally with an atrial septal defect or as an isolated lesion. Very rarely it occurs with tricuspid atresia, intact ventricular septum, and dysplasia of the right ventricular free wall and of the ventricular septum. We present the clinical, anatomic, and histologic findings of a new case, and for the first time, the data from two patients with absent pulmonary valve and severe tricuspid stenosis, who exhibited similar histologic findings. We also reviewed the clinical and anatomic data of 24 previously published cases and compared them with the new cases. In all three new cases, the myocardium of the right ventricle was very abnormal. In the two cases with tricuspid stenosis, large segments of myocardium were replaced with sinusoids and fibrous tissue. In the case with tricuspid atresia, the right ventricular free wall contained only fibroelastic tissue. The ventricular septum in all three patients showed asymmetric hypertrophy and in two of the three patients, multiple sinusoids had replaced large segments of myocardial cells. The left ventricular free wall myocardium and the walls of the great arteries were unremarkable. Our data indicate that myocardial depletion involving the right ventricular free wall and the ventricular septum and its replacement by sinusoids and fibroelastic tissue occur not only in cases of absent pulmonary valve with tricuspid atresia but also in cases of absent pulmonary valve with tricuspid stenosis. The degree of myocardial depletion varies and is more severe when the tricuspid valve is atretic.
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2/13. Resolution of protein-losing enteropathy with standard high molecular heparin and urokinase after Fontan repair in a patient with tricuspid atresia.

    At 6 years of age, a girl with tricuspid atresia underwent a Bjork modified fontan procedure with implantation of a Carpentier Edwards bioprosthesis between the right atrium and the right ventricle. Ten years later she developed increasing edema, ascites and pleural effusions. The work-up showed severe stenosis of the bioprosthesis and protein losing enteropathy with a massive decrease of the albumin level to 14 g/l (normal 40-50 g/l). At 17 years of age, the bioprosthesis was replaced with a direct anastomosis between the cavoatrial junction and the right pulmonary artery. Within one month post-operatively, extensive thrombosis of the superior vena cava, anonymous and subclavian veins occurred. Protein-losing enteropathy persisted with an albumin level of 17 g/l. Parallel to the successful treatment of these thrombi with high molecular heparin and urokinase, protein losing enteropathy and hypoalbuminemia resolved completely as long as the antithrombotic treatment with high molecular heparin was continued. Oral anticoagulation was ineffective. Chronic antithrombotic treatment with high molecular heparin may thus be the treatment of choice in these forms of protein-losing enteropathy associated with venous thrombosis.
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3/13. Absorbable pulmonary artery banding in tricuspid atresia.

    A child with tricuspid atresia, concordant ventriculoarterial connections, large ventricular septal defect, and elevated pulmonary artery pressure underwent pulmonary artery banding with a polydioxanone ribbon. This procedure was successful in this patient as the ventricular septal defect became restrictive while the banding was fully absorbed after 5 months. This technique could be included in the panel of surgical strategies for patients with single ventricle physiology and potential but insufficient subpulmonary stenosis in early infancy.
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4/13. Double-outlet left ventricle. Echocardiographic diagnosis.

    This is a case report of a double-outlet left ventricle associated with tricuspid atresia and hypoplasia of the right ventricle, diagnosed during echocardiography with color-flow imaging, in a three-month-old child who presented with fatigue and cyanosis. The child underwent palliative pulmonary arterial banding without an invasive procedure, and showed sustained improvement during follow-up.
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5/13. prenatal diagnosis of isolated tricuspid valve atresia: report of 4 cases and review of the literature.

    OBJECTIVE: To describe the prenatal features of fetal tricuspid atresia. methods: Four cases of fetal tricuspid atresia were prenatally diagnosed, sonographically described, and followed. RESULTS: On the basis of this small series, the key findings for diagnosis included the demonstration of no patent tricuspid valve on the 4-chamber view, no flow across the tricuspid valve on pulsed or color Doppler flow mapping, small right ventricles, and associated interventricular septal defects. Increased nuchal translucency thickness may give the first clue leading to follow-up scans, resulting in a definite diagnosis. CONCLUSIONS: tricuspid atresia can be readily diagnosed prenatally. The key findings and differential diagnoses are provided.
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6/13. Successful transcatheter coil embolization of coronary artery to left ventricular fistula associated with absent pulmonary valve with tricuspid atresia in early infancy.

    Transcatheter coil embolization for coronary artery to left ventricular fistula was successfully performed in a neonate. At 30 weeks' gestation, fetal echocardiography showed a hypoplastic right ventricle with intact ventricular septum, absent pulmonary valve, tricuspid atresia, and marked distension of the right coronary artery. After birth, the neonate had congestive heart failure and the electrocardiogram showed myocardial ischemic changes in the left ventricular area. aortography showed a dilated right coronary artery arising from the ascending aorta and draining into the left ventricle. Transcatheter coil embolization was carried out on the 9th day after birth. Since the procedure, no myocardial ischemic changes have been detected. Transcatheter coil embolization is a useful therapy for coronary artery fistula associated with myocardial ischemia.
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7/13. Multiple fistulas from the coronary arteries to the left ventricle in tricuspid atresia.

    It is rare to find multiple fistulas arising from all three coronary arteries and draining into the left ventricle. coronary angiography revealed this anomaly in a one-year-old girl with tricuspid atresia after conversion to the Fontan circulation. To the best of our knowledge, this is the first report of such multiple fistulas in the setting of tricuspid atresia, and also the first report in childhood.
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8/13. Performance of the fontan procedure using extra-cardiac direct anastomosis on patients treated for penicillin-resistant streptococcus pneumoniae endocarditis.

    streptococcus pneumoniae is now a rare cause of endocarditis in humans. We report a patient with a double outlet right ventricle and mitral atresia, who underwent the fontan procedure without prosthetic materials after treatment for penicillin-resistant streptococcus pneumoniae endocarditis. Postoperative infectious endocarditis was not found. In patients with a history of infectious endocarditis, direct anastomosis of the main pulmonary artery and inferior vena cava would reduce the risk of recurrent infectious endocarditis.
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9/13. Reversal of fenestration flow during ventricular systole in Fontan patients in junctional or ventricular paced rhythm.

    Sinus node dysfunction is relatively common in patients with Fontan palliation for single ventricle congenital heart disease, and such patients often are in junctional rhythm or may have pacemaker systems for bradycardia. Because the physiologic determinants of left atrial pressure play a major role in determining pulmonary blood flow and therefore cardiac output in Fontan patients, the loss of atrioventricular (AV) synchrony in junctional rhythm or demand ventricular pacing in these patients might be expected to influence cardiac output. We report two cases of Fontan patients with the absence of AV synchrony that resulted in reversal of flow through the Fontan fenestration during ventricular systole. In both cases, restoration of AV synchrony by atrial pacing resulted in the elimination of retrograde fenestration flow, increased cardiac output, and improved clinical status.
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10/13. Percutaneously adjustable pulmonary artery band.

    BACKGROUND. pulmonary artery banding, originally introduced to palliate increased pulmonary blood flow, is now primarily used for complex congenital cardiac defects with a prohibitive risk in the early repair, such as tricuspid atresia, or occasionally to prepare the left ventricle for eventual arterial switch. Conventional banding is, however, often poorly tolerated, and the precise adjustment required to reduced pulmonary blood flow or sufficiently challenge the left ventricle is difficult and can cause life-threatening hemodynamic changes. methods. We designed a band that allows accurate tightening and is reversible during the operation and at the bedside with the chest closed, allowing precise gradual adjustment over days, thereby minimizing cardiovascular instability. This design is the extension of previous work done by one of us on an adjustable annuloplasty ring. RESULTS. The band has been used for a 16-month-old child with tricuspid atresia type 1C who had undergone two failed attempts at banding. Hemodynamic measurements after 3 months confirmed achievement of the desired pulmonary pressure. CONCLUSIONS. This design, the simple insertion, and the ability to finely readjust the banding over days, combined with excellent patient tolerance, encourage further use of our adjustable band.
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