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Cor triatriatum dexter: A rare cause of cyanosis during neonatal period.

ABSTRACT: Cor-triatriatum dexter is an extremely rare congenital heart defect in which there is complete persistence of the right valve of embryonic sinus venosus that results in partitioning of the right atrium into a smooth and trabeculated portion. The smooth portion receives venous blood from inferior vena cava, superior vena cava, and coronary sinus while the trabeculated portion contains the right atrial appendage and the opening of tricuspid valve. We report a 1-week-old child who presented with intermittent episodes of central cyanosis. Echocardiography, established, and bubble contrast study confirmed the diagnosis of an isolated cor-triatriatum dexter. The baby initially underwent an intervention by cardiac catheterization, which was unsuccessful in disrupting the membrane and re-direct the systemic venous flow to the right heart chambers. She subsequently had the cor-triatriatum dexter membrane resected via an uncomplicated open-heart surgery.

SUBMITTER: Alghamdi MH

PROVIDER: S-EPMC4782468 | biostudies-literature | 2016 Jan-Apr

REPOSITORIES: biostudies-literature

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Cor triatriatum dexter: A rare cause of cyanosis during neonatal period.

Alghamdi Mohammed Hussien MH

Annals of pediatric cardiology 20160101 1

Cor-triatriatum dexter is an extremely rare congenital heart defect in which there is complete persistence of the right valve of embryonic sinus venosus that results in partitioning of the right atrium into a smooth and trabeculated portion. The smooth portion receives venous blood from inferior vena cava, superior vena cava, and coronary sinus while the trabeculated portion contains the right atrial appendage and the opening of tricuspid valve. We report a 1-week-old child who presented with in ...[more]

PMID: 27011692

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Cor Triatriatum Dexter: Contrast Echocardiography Is Key to the Diagnosis of a Rare but Treatable Cause of Neonatal Persistent Cyanosis.

Project description:Cor triatriatum dexter (CTD) is an extremely uncommon and underreported congenital cardiac anomaly in which the persistence of the embryonic right venous valve separates the right atrium into two chambers with varying degrees of obstruction to antegrade flow and variable degree of right to left shunt at atrial level. Depending on the size of the valves, clinical manifestations vary from absence of symptoms to severe hypoxia, requiring urgent surgical correction. We herein describe the diagnostic difficulties in a case of neonatal CTD, who developed increasingly severe and unresponsive cyanosis, first interpreted as postnatal maladjustment with pulmonary hypertension. The failure to respond to oxygen and pulmonary vasodilators led us to reconsider a different diagnosis. The use of contrast echocardiography improved the diagnostic performance of transthoracic echocardiogram (TTE) and revealed a massive right-to-left shunt secondary to the presence of an atrial membrane that required urgent surgery.

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Percutaneous Balloon Dilation in Two Dogs with Cor Triatriatum Dexter.

Project description:Percutaneous balloon dilation was performed in a Rhodesian Ridgeback and in an American Staffordshire Terrier affected by cor triatriatum dexter (CTD). Both cases had ascites without jugular venous distension or pleural effusion. In both dogs the CTD presented a perforated membrane but with different morphology: in one case the coronary sinus entered the caudal chamber of the CTD together with the caudal vena cava. In the other case, the coronary sinus communicated with the cranial chamber of the CTD together with the cranial vena cava. Percutaneous balloon dilation of the CTD was successfully performed, and both dogs had uneventful surgery recoveries. At two years of follow-up, the dogs were free from clinical signs and cardiac medication.

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Transcatheter balloon dilatation of cor triatriatum dexter with percutaneous atrial septal defect closure.

Project description:Cor triatriatum dexter is an extremely rare congenital heart defect and cause of hypoxia in adults. We describe a case of cor triatriatum dexter discovered incidentally due to an iatrogenic atrial septal defect. The cor triatriatum dexter resolved with balloon dilation - a novel technique to manage this rare clinical condition. <Learning objective: Management of cor triatriatum dexter, an exceptionally rare diagnosis, has often been thought to be surgical. However, percutaneous intervention may be considered in select clinical situations.>.

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Wong's anomaly - a rare variant of cor triatriatum.

Project description:Cor triatriatum sinistrum is an extremely rare congenital heart disease. It is even more uncommon in adults, and clinically significant mitral valve lesion complicating cor triatriatum is distinctly rare. Wong et al reported for the first time the rare combination of cor triatriatum sinister associated with severe mitral regurgitation and abnormal tensor apparatus of the mitral valve. We report a similar case and used the term Wong's anomaly for the syndrome, having membranous type of cor triatriatum sinistrum, severe mitral regurgitation and hypoplasia of the papillary muscles and short chordae. Color Doppler Echocardiography showed peculiar 'helmet sign' of mitral regurgitation, wherein the mitral regurgitation color jet fills the distal atrial chamber and abruptly ends in a horizontal plane as it is halted by the intra-atrial membrane.

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Percutaneous Edge-to-Edge Tricuspid Valve Repair in a Patient with Cor Triatriatum Dexter: A Case Report.

Project description:BackgroundTricuspid regurgitation is gaining importance due to its high morbidity and mortality. Especially in the elderly, novel technologies in percutaneous therapies have become valuable options due to the commonly present high surgical risk.Case presentationWe report a case of a 78-year-old female suffering from massive tricuspid regurgitation with repetitive right-sided heart failure hospitalizations. As the patient was very frail and deemed as high surgical risk, we used the TriClip® system to improve her symptomatic status. During diagnostic work-up, an additional membrane separating the right atrium, consistent with the definition of a cor triatriatum dexter, was found. Although increasing the complexity of the procedure, implantation of 3 clips with reduction of tricuspid regurgitation to a mild-to-moderate degree was achieved without any notable complications. The patient was discharged with ameliorated symptoms on the fourth postoperative day.ConclusionsOur case highlights the feasibility of percutaneous edge-to-edge tricuspid valve repair in an elderly woman with cor triatriatum dexter. Accurate echocardiographic visualization is an absolute requirement to gain access to the tricuspid valve without interacting with prevailing additional membranes.

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Cor Triatriatum: A Tale of Two Membranes.

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A rare case of partial cor triatriatum diagnosed at an adult age: a case report.

Project description:BackgroundCor triatriatum sinistra (CTS) is a rare condition where the left atrium (LA) is divided by a thin membrane into an upper and lower chamber. Incidentally, the diagnosis is made in late adulthood, usually because of a favourable variant such as in our patient who presented with partial CTS.Case summaryWe present the case of a 62-year-old female who presented with COVID-19. She was known for longstanding symptoms of dyspnoea on exertion as well as a minor stroke several years ago. Computed tomography on admission suggested there was a mass in the LA but transthoracic echocardiography and cardiac magnetic resonance imaging revealed the diagnosis of partial CTS in which the superior compartment received pulmonary venous drainage from the right lung and the left-sided pulmonary veins drained into the inferior chamber. Since there were signs of chronic pulmonary oedema she successfully underwent balloon dilatation of the membrane resulting in remission of symptoms and normalization of the pressure in the accessory chamber.DiscussionPartial CTS is a rare variant of CTS. Since part of the pulmonary veins drains in the lower chamber of the LA (and thereby unload the right ventricle), it is a favourable variant and patients may present later in life when membrane orifices calcify or it may be discovered as an incidental finding. In some patients requiring intervention, balloon dilatation of the membrane may be considered as an alternative to surgical removal of the membrane by thoracotomy.

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