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We report 3 cases of cor triatriatum that were diagnosed late, in the 4th and 5th decades of life. The presentations of these 2 men and 1 woman varied substantially, both in anatomic and symptomatic aspects. The woman had an associated complex congenital anomaly, which is not uncommon in cases of cor triatriatum. We present our cases, along with a review of this rare congenital cardiac anomaly.
Twenty-five patients with cor triatriatum underwent surgical correction at the Texas Heart Institute during a 21-year period from 1959 to 1980. Patients ranged in age from 4 months to 38 years. Diagnosis was established preoperatively in 14 patients (56%), at the time of operation for correction of associated lesions in 10 patients (40%), and during reoperation in 1 (4%). In the earlier part of this series, diagnosis was more common at the time of operation. Associated cardiovascular anomalies were present in 20 patients (80%), major anomalies in 14 others (56%). Simultaneous correction or palliation of associated lesions and total excision of the anomalous membrane were done in 18 patients utilizing temporary cardiopulmonary bypass. Cor triatriatum alone was repaired in 5 patients (20%) with the aid of extracorporeal circulation. Two patients underwent reoperation: 1 because of incomplete excision of the septum and the other because the condition had not been diagnosed during a first operation for correction of total anomalous pulmonary venous return. Excision of the membrane was accomplished utilizing the left atrium in 10 patients (40%), the right atrium in 12 (48%), or both in 3 (12%). Four patients (16%) died early after operation; all were infants who had severe associated cardiac anomalies. Among the 21 surviving patients, results were excellent in 20.
Successful repair of the rare anomaly of concomitant cor triatriatum and coronary sinus total anomalous pulmonary venous connection in a neonate is described. The presence of pulmonary venous obstruction or cardiomegaly, or both, in a child with total anomalous pulmonary venous connection should alert to the possibility of a coexistent cor triatriatum.
Classic cor triatriatum is rarely found in adults. Preoperative assessment of classic cor triatriatum in a 22 year old man without symptoms by three dimensional echocardiography was more informative than transoesophageal echocardiography or magnetic resonance imaging, which both showed only a small hole in the membrane separating the accessory atrium from the true left atrium. The size of the hole indicated a strong likelihood that symptoms would develop in this patient. Dynamic three dimensional echocardiography, however, showed a long wide hole in the separating membrane. This finding was consistent with the absence of symptoms in this patient. Symptom free patients with moderate obstruction do not need early surgical correction.
Surgical correction of cor triatriatum with unroofed coronary sinus consisted of creation of the coronary sinus using the left atrial diaphragm and closure of the atrial septal defect in an adult patient. No materials other than intracardiac components were used to repair all anomalies.
BACKGROUND: Cor triatriatum is rarely found in adults and its diagnosis is mostly dependent on transesophageal echocardiogram. We present a case of an adult male with cor triatriatum and discuss our approach to diagnosis with a review of literature. CASE: A 49-year-old male presented with progressive exertional dyspnea for the past two-and-half years. A transthoracic echocardiogram revealed a thin linear echo-dense structure traversing the left atrium (LA). A transesophageal echocardiogram demonstrated a thin immobile membrane in the LA attached medially to the interatrial septum. No fenestration of the membrane was seen. A swirling spontaneous contrast was noted in the posterior chamber. Color Doppler did not reveal any flow across the membrane. A CT scan of the chest was unhelpful to differentiate extracardiac versus intracardiac origin. Contrast echocardiogram with Optison was performed, which showed a differential opacification of the two atrial chambers and delayed emptying of contrast into true LA establishing communication between the chambers. Left and right heart catheterization were performed with simultaneous pulmonary capillary wedge and left ventricular end diastolic pressure measurement, revealing a mean gradient of 17 mmHg. Surgical correction of the membrane was recommended. Intraoperatively, an orifice of 0.7 cm was identified in an eccentric position with mosaic pattern of continuous turbulent flow across the membrane by color Doppler. DISCUSSION: Our case demonstrates the impact of contrast echocardiography in a situation where the transesophageal approaches were limited even with the aid of color Doppler.
Cor triatriatum presenting in adulthood is extremely rare. An 18-year-old female patient presented with a clinical picture masquerading mitral valvular heart disease. Both transthoracic and transesophageal echocardiography demonstrated a membrane in the left atrium with no other cardiac anomaly. The patient was successfully operated on without the need for catheterization.
A case of subtotal cor triatriatum associated with a partial anomalous pulmonary venous drainage of the left lung, in a 30 months old boy is reported. Diagnosis was suggested by the presence of pulmonary venous obstruction and pulmonary hypertension. Doppler, echocardiogram diagnosed the cor triatriatum and the degree of the pulmonary hypertension. The cine-angiography demonstrated the anomalous, obstructive, left pulmonary venous drainage. The pulmonary hypertension has mandatorised an early surgical cure, the result was satisfactory at 16 months.
Cor triatrium sinister is a rare congenital disease (0,1% of cases of congenital heart disease). Cor triatrium is recognized by the finding of an abnormal fibromuscular membrane that subdivise the left atrium into posterosuperior and anteroinferor chambers. This anomaly creates an obstacle to the venous pulmonary flow. Depending on the severity of obstruction, cor triatrium may be symptomatic in childhood but it can be symtom free even in adulthood. Diagnosis of this abnormality has been easy supported by transthoracic and transoesophageal echocardiography.Treatment is easy to perform and consists in surgical defenitive correction in symptomatic patients. We report a case of a 31-year-old man in whom a non obstructive cor triatrium was discovered while evaluation for an atrial fibrillation secondary to a hyperthyroidism. According to this rare case we try to evaluate the severity and the outcome of this disease relating the literature data.
An 80-year-old man was admitted to the hospital because of recurrent syncope caused by sick sinus syndrome. Conventional transthoracic echocardiography showed a membranelike echo-dense structure across the left atrium. Subsequently we confirmed the diagnosis of cor triatriatum with multiple defects and shunt flows across the membrane by transesophageal color flow Doppler echocardiography. There were no other associated congenital anomalies. The patient recovered well and was discharged from the hospital after implantation of a permanent VVI pacemaker for sinus node dysfunction. No other cardiac intervention for the cor triatriatum was done except follow-up with echocardiography. About 1 year after discharge, he died of massive cerebral infarction. The cardiac autopsy findings were compatible with the transesophageal echocardiogram. This is a rare case of uncorrected cor triatriatum in an elderly patient.
The diagnosis of cor triatriatum in an adult was made from routine two-dimensional transthoracic echocardiography. The findings of aliasing and turbulence in the roof of the left atrium suggested pulmonary venous stenosis. A transesophageal echocardiogram defined both the hemodynamic features of nonobstructing cor triatriatum and the presence of isolated pulmonary venous stenosis. The clinical use of transesophageal echocardiography with color flow Doppler in the elucidation of complex anatomic substrate is demonstrated.
A shiba inu dog with marked abdominal distension was diagnosed with cor triatriatum dexter and surgical correction was performed under extracorporeal circulation. The total duration of cardiac arrest was 11 minutes and total perfusion time was 34 minutes. The dog had an uneventful postoperative recovery. Postoperative contrast radiography of the caudal vena cava revealed normal flow into the right heart. Abdominal distension was no longer observed. Although several methods have been used to treat cor triatriatum dexter in dogs, the authors consider surgical correction under extracorporeal circulation to be a reliable approach.
A surgical treatment of a 54 year-old female with cor triatriatum was reported. Two dimensional echocardiography had demonstrated an abnormal septum in the left atrium. The abnormal septum had a 2 x 1 cm fenestration and existed between the left and right pulmonary veins. Preoperatively, we had misjudged the septum as ASD and misdiagnosed this case as PAPVC. During surgery we found that there was the accessory atrial chamber posterior to the true ASD. The accessory chamber received right pulmonary veins and connected to the left atrium. The left pulmonary veins connected to the left atrium normally behind the abnormal septum. We diagnosed this case as Lucas-Shmidt IIIA1 type cor triatriatum with ASD. The abnormal septum was resected, and the ASD was closed with a bovine pericardium. The postoperative course was uneventful and she was discharged 19 days after the operation.
Cor triatriatum and imperforate anus are rare. We describe, for the first time, the coexistence of these conditions in a 43 year old woman.
We report on an asymptomatic 2 1/4 year old girl. Echocardiography revealed cor triatriatum with a membrane separating the left atrium. Transesophageal echocardiography demonstrated 3 large perforations of the intraatrial membrane. Using Doppler sonography, no pressure gradients across these orifices were identified. Obstruction of the intraatrial membrane and pulmonary hypertension were excluded by cardiac catheterization. We therefore decided against surgical resection of the membrane and to follow the patient noninvasively by echocardiography. Follow-up over 18 months revealed no development of an obstruction across the intraatrial membrane. Our case shows that immediate surgical correction is not necessary in all patients with cor triatriatum. Conservative management of these patients requires full evaluation of the hemodynamics and careful follow-up examinations by echocardiography.
Cor triatriatum dexter is an unusual cardiac abnormality with division between the sinus and primitive atrial portions of the right atrium. Three-dimensional echocardiography is a novel technique that defines this entity.
A patient with rheumatic mitral stenosis associated with cor triatriatum is described. The anomalies were detected by two-dimensional echocardiography and confirmed by transesophageal echocardiography. Percutaneous mitral valvuloplasty was successfully performed with the inoue technique. The clinical and technical implications during the procedure of this previously unreported association are discussed.
A case report of a variant type of cor triatriatum was presented. The patient was critically ill when she was admitted to our hospital. An emergent operation was performed under the cardio-pulmonary bypass. The right atrium was incised, then the interatrial septum was incised. Inspection revealed an abnormal membrane in the left atrium. All the pulmonary veins were drained into the accessory chamber. The left atrial appendage was also connected with the accessory chamber. The membrane was not including the mitral valve tissue. It was resected completely. Postoperative course was uneventful.