Cor Triatriatum

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Background

First reported in 1868,[1] cor triatriatum, that is, a heart with 3 atria (triatrial heart), is a congenital anomaly in which the left atrium (cor triatriatum sinistrum) or right atrium (cor triatriatum dextrum) is divided into 2 compartments by a fold of tissue, a membrane, or a fibromuscular band.[2, 3, 4, 5, 6, 7] Classically, the proximal (upper or superior) portion of the corresponding atrium receives venous blood, whereas the distal (lower or inferior) portion is in contact with the atrioventricular valve and contains the atrial appendage and the true atrial septum that bears the fossa ovalis. The membrane that separates the atrium into 2 parts varies significantly in size and shape. It may appear similar to a diaphragm or be funnel-shaped, bandlike, entirely intact (imperforate) or contain 1 or more openings (fenestrations) ranging from small, restrictive-type to large and widely open. See the images below.


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Cor triatriatum. Echocardiogram showing the proximal chamber (PC) and distal chamber (DC) of the left atrium; the right atrium (RA), left ventricle (L....


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Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Image

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

In the pediatric population, this anomaly may be associated with major congenital cardiac lesions such as tetralogy of Fallot, double outlet right ventricle, coarctation of the aorta, partial anomalous pulmonary venous connection, persistent left superior vena cava with unroofed coronary sinus, ventricular septal defect, atrioventricular septal (endocardial cushion) defect, and common atrioventricular canal.[8, 9, 10] Rarely, asplenia or polysplenia has been reported in these patients. Although frequently an isolated finding,[11, 12, 13, 14, 15, 16, 17, 18] cor triatriatum in the adult has been reported in association with ostium secundum atrial septal defect, dilated coronary sinus due to persistent left superior vena cava, and bicuspid aortic valve.[19]

Cor triatriatum dextrum is extremely rare and results from the complete persistence of the right sinus valve of the embryonic heart. The membrane divides the right atrium into a proximal (upper) and a distal (lower) chamber. The upper chamber receives the venous blood from both vena cavae and the lower chamber is in contact with the tricuspid valve and the right atrial appendage.

Pathophysiology

Cor triatriatum sinistrum

The most popular theory holds that cor triatriatum sinister occurs when the common pulmonary vein fails to incorporate the pulmonary circulation into the left atrium and the common pulmonary venous ostium remains narrow (malincorporation theory). The result is a septum-like structure that divides the left atrium into 2 compartments. However, this theory fails to explain the presence of fossa ovalis and atrial muscle fibers within the walls of the proximal chamber where only a venous wall is supposed to be present.[20, 21, 22, 23, 24, 25, 26, 27, 28]

In addition, several cases have been reported in which 1 or 2 pulmonary veins drain into the proximal (accessory) chamber and the others drain directly into the true left atrium. Others believe that the membrane dividing the left atrium is an abnormal growth of the septum primum (malseptation theory) or that the right horn of the embryonic sinus venosus entraps the common pulmonary vein and thereby prevents its incorporation into the left atrium (entrapment theory). The significance of a prominent or persistent left superior vena cava in the pathogenesis of cor triatriatum is unclear.

Cor triatriatum dextrum

During embryogenesis, the right horn of the sinus venosus gradually incorporates into the right atrium to form the smooth posterior portion of the right atrium, whereas the original embryologic right atrium forms the trabeculated anterior portion. The right horn of the sinus venosus and the embryologic right atrium are then connected through the sinoatrial orifice, which has on either side the 2 valvular folds called the right and left venous valves. During this incorporation, the right valve of the right horn of the sinus venosus divides the right atrium in 2. This right valve forms a sheet that serves to direct the oxygenated venous return from the inferior vena cava across the foramen ovale to the left side of the heart during fetal life (Chiari network).[29, 30, 31, 32, 33, 34, 35, 36]

Normally, this network regresses and leaves behind the crista terminalis superiorly and the eustachian valve of the inferior vena cava and the thebesian valve of the coronary sinus inferiorly. Complete persistence of the right sinus valve of embryonic life results in separation of the smooth and trabeculated portions of the right atrium and constitutes cor triatriatum dextrum. If this membrane is extensively fenestrated and weblike in appearance, then it is referred to as the Chiari network.

Epidemiology

Frequency

United States

Mortality/Morbidity

Sex

No clear difference has been noted in incidence or clinical presentation among men or women.

Age

History

Cor triatriatum sinistrum

Cor triatriatum dextrum

Physical

Cor triatriatum sinistrum

Cor triatriatum dextrum

Laboratory Studies

Cor triatriatum sinistrum

Cor triatriatum dextrum

There are no pathognomonic electrocardiographic findings in isolated cor triatriatum dextrum. Right heart catheterization may reveal elevated pressure in the proximal right atrial chamber with a gradient across the accessory membrane.

Imaging Studies

Cor triatriatum sinistrum

Frequently reported associated findings on cardiac imaging include the following:

Cor triatriatum dextrum

In general, advanced cardiac imaging (transthoracic and transesophageal echocardiography, cardiac MRI, and right ventriculography) demonstrates the presence of a membrane within the right atrium and may also be diagnostic of other concomitant congenital or acquired cardiac abnormalities.[40, 53, 41, 42, 43, 57, 44, 45, 58, 46, 47, 48, 59, 50, 54, 60]

See the videos below for imaging studies of cor triatriatum.


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Cor triatriatum. This film shows the classic pattern of pulmonary edema associated with pulmonary overcirculation and pulmonary venous obstruction. Patient has an anomalous pulmonary venous connection that was only obvious after a pulmonary artery shunt. The particular radiograph is not a patient with cor triatriatum, but appearance of prominent pulmonary vascularity is the same. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Video

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Video

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Video

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Video

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.


View Video

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Medical Care

Medial care for patients with cor triatriatum includes the following:

Surgical Care

Consultations

Diet

Activity

Medication Summary

The medical management of cor triatriatum is targeted towards the associated elevation in pulmonary vascular resistance and heart failure. It is continued in the postoperative period until resistance falls and right ventricular performance improves. The mainstays of treatment are inotropic agents and diuretics.

Digoxin (Lanoxin)

Clinical Context:  Exerts inotropic action by increasing amount of intracellular calcium available during excitation-contraction coupling. One of numerous inotropic agents used in infants with congenital cardiac defects. Other agents, such as dopamine, are more appropriate for the acute management of heart failure in the ICU setting.

Dopamine (Intropin)

Clinical Context:  Adrenergic agonists often are used in the critical care setting for rapid onset of action and rapid time to peak effect. Are much easier to titrate to effect in acute setting. Half-life is much shorter than digoxin, and effects are rapidly lost when drug is discontinued.

Class Summary

Provide myocardial support in the perioperative period for patients with right heart failure. The more restrictive the connection between the proximal and distal chambers, the more likely inotropic support will be required. A number of agents are available in this category. Adrenergic agonists increase myocardial contractility in patients with heart failure.

Furosemide (Lasix)

Clinical Context:  Highly effective first-line drug for diuresis in newborns and infants. Sulfonamide derivative that exerts effects on loop of Henle and distal renal tubule, inhibiting reabsorption of sodium and chloride.

Class Summary

Management of right heart failure and pulmonary edema.

Heparin

Clinical Context:  Augments activity of antithrombin III and prevents conversion of fibrinogen to fibrin. Does not actively lyse but is able to inhibit further thrombogenesis. Prevents reaccumulation of clot after spontaneous fibrinolysis.

Warfarin (Coumadin)

Clinical Context:  Most commonly used oral anticoagulant. Interferes with hepatic synthesis of vitamin K-dependent coagulation factors; used for prophylaxis and treatment of thromboembolic disorders.

Class Summary

These agents are used in the prophylaxis and treatment of thromboembolic disorders.

Further Inpatient Care

Further inpatient care of patients with cor triatriatum includes routine postoperative surgical wound care and management of postoperative arrhythmias.

Transfer

Symptomatic patients may have to be transferred to tertiary care centers for advanced cardiac imaging, angiographic evaluation, and surgical correction.

Complications

Prognosis

Author

Jamshid Shirani, MD, Director of Cardiology Fellowship Program, Director of Echocardiography Laboratory, St Luke's University Health Network

Disclosure: Nothing to disclose.

Coauthor(s)

Arun Kalyanasundaram, MD, MPH, Interventional Cardiology Fellow, Department of Cardiology, Cleveland Clinic

Disclosure: Nothing to disclose.

Kamal K Pourmoghadam, MD, Associate Professor, Department of Cardiothoracic Surgery, Jefferson Medical College; Director of Pediatric Cardiac Surgery, Department of Surgery, Janet Weis Children's Hospital, Geisinger Medical Center

Disclosure: Nothing to disclose.

Yuba R Acharya, MD, Fellow, Department of Cardiology, St Luke's University Hospital, Bethlehem, Pennsylvania

Disclosure: Nothing to disclose.

Specialty Editors

Francisco Talavera, PharmD, PhD, Adjunct Assistant Professor, University of Nebraska Medical Center College of Pharmacy; Editor-in-Chief, Medscape Drug Reference

Disclosure: Medscape Salary Employment

Frank M Sheridan, MD, Cardiology, Providence Everett Medical Center

Disclosure: Nothing to disclose.

Amer Suleman, MD, Private Practice

Disclosure: Nothing to disclose.

Chief Editor

Park W Willis IV, MD, Sarah Graham Distinguished Professor of Medicine and Pediatrics, University of North Carolina at Chapel Hill School of Medicine

Disclosure: Nothing to disclose.

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Cor triatriatum. Echocardiogram showing the proximal chamber (PC) and distal chamber (DC) of the left atrium; the right atrium (RA), left ventricle (LV), and right ventricle (RV) also are shown. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. This film shows the classic pattern of pulmonary edema associated with pulmonary overcirculation and pulmonary venous obstruction. Patient has an anomalous pulmonary venous connection that was only obvious after a pulmonary artery shunt. The particular radiograph is not a patient with cor triatriatum, but appearance of prominent pulmonary vascularity is the same. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Echocardiogram showing the proximal chamber (PC) and distal chamber (DC) of the left atrium; the right atrium (RA), left ventricle (LV), and right ventricle (RV) also are shown. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Image courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. This film shows the classic pattern of pulmonary edema associated with pulmonary overcirculation and pulmonary venous obstruction. Patient has an anomalous pulmonary venous connection that was only obvious after a pulmonary artery shunt. The particular radiograph is not a patient with cor triatriatum, but appearance of prominent pulmonary vascularity is the same. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.

Cor triatriatum. Movie courtesy of Guido Giordano, MD, Cardiovascular Department, Azienda Ospedaliera Cannizzaro, Catania, Italy.