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Ann Thorac Surg 2005;80:1666-1671
© 2005 The Society of Thoracic Surgeons

Original article: Cardiovascular

Cor Triatriatum: Presentation, Diagnosis and Long-Term Surgical Results

Nelson Alphonso, MD * , Martin A. Nørgaard, MD, Andrew Newcomb, MD, Yves d'Udekem, MD, Christian P. Brizard, MD, Andrew Cochrane, MD

Victorian Pediatric Cardiac Surgery Unit, Royal Children's Hospital, Melbourne, Australia

Accepted for publication April 27, 2005.

* Address correspondence to Dr Alphonso, UCSF Department of Surgery, Division of Pediatric Cardiothoracic Surgery, 513 Parnassus Ave, Room S-549, Box 0117, San Francisco, CA 94143-0117 (Email: nelson.a{at}bigfoot.com).


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Cor triatriatum sinister is a rare, surgically correctable, congenital cardiac anomaly. Fewer than 250 cases have been reported. It can occur as an isolated defect (classic) or in association with other congenital cardiac anomalies (atypical).

METHODS: A retrospective review of 28 consecutive patients diagnosed at the Royal Children's Hospital in Melbourne, Australia during a 22-year span from 1981 to 2003.

RESULTS: There were 13 men (46%) and 15 women (54%). Fifteen patients were less than 1 year of age (neonates, [n = 7], 25%; infants, [n = 8], 29%). 17 patients (61%) had a communication between the right atrium and either the proximal or distal chamber. Fifteen patients (54%) had atypical cor triatriatum. Median age at presentation was 6 months (range, 0.6 to 240). Twenty-four patients (86%) had presented by 5 years of age. Five patients (18%) underwent emergency surgery. Median age at operation was 7 months (range, 1 to 243). Twenty-seven patients (96%) underwent preoperative transthoracic echocardiography. Nine patients (32%), all with atypical cor triatriatum, required cardiac catheterization for diagnosis. Twenty-seven patients (96%) were correctly diagnosed before treatment. Twenty-seven patients (96%) were treated surgically. The defect was approached through the right atrium in 26 patients (93%). There was 1 early death and 1 patient died 10 years after repair. Follow-up was 86% complete. At a median follow-up of 98 months (range, 0.2 to 284), all patients including those with atypical cor triatriatum were in New York Heart Association's function class 1. Post-repair survival was 96% and 88% at 5 and 15 years, respectively.

CONCLUSIONS: Surgery offers good early and long-term results for both classic and atypical cor triatriatum.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 References
 
Cor triatriatum sinister is a rare but surgically correctable congenital cardiac anomaly accounting for 0.1% of all congenital cardiac malformations [1]. In cor triatriatum the pulmonary veins enter a postero-superior proximal left atrial chamber that is separated from the antero-inferior distal left atrial chamber containing the mitral valve and left atrial appendage by a diaphragm in which there are one or more restrictive ostia. It is associated with other congenital cardiac anomalies (atypical type) in 24% to 80% of cases (Table 1).


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  		Table 1. Results of Surgical Series of Cor Triatriatum Since 1981
 
Church [2] first described cor triatriatum in 1868. Lewis and colleagues [3] and Vineberg and Gialloreto [4] reported the first surgical repair in 1956. Lewis and colleagues [3] incised the diaphragm using hypothermia and inflow occlusion and Vineberg used finger fracture through the left atrial appendage. Fewer than 250 cases have since been reported in the literature [5–11]. We reviewed our experience with cor triatriatum over a 22-year period between April 1981 and June 2003 with respect to morphology, presenting symptoms, diagnosis, operative results, and long-term outcome. We believe that this represents the largest series of patients diagnosed with cor triatriatum.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
Patient Population
Between April 1981 and October 2003, 28 patients with cor triatriatum sinister presented to our institution. Twenty-seven patients (96%) required surgical correction. Eight consultants undertook the surgical treatment during this period, and the technique used was at the discretion of the surgeon. The presentation, echocardiographic and cardiac catheterization data, operative notes, and postoperative course of each patient were retrospectively reviewed.

Morphology
For an accurate description of the findings we have used the classification system first described by Lam and colleagues [12] in 1962 (Table 2).


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  		Table 2. Lam Classification of Cor Triatriatum
 
Timing of Operation
The timing of operation reflected the severity of symptoms. Patients with insidious symptoms were operated on electively, whereas those with more severe symptoms were operated within the same hospital admission.

Outcome
Early death was defined as death occurring during the hospital admission for operation or within 30 days after surgery if discharged earlier. All other deaths were classified as late deaths.

Follow-Up
All follow-ups were obtained during the period of study. Patients were contacted by telephone regarding symptoms, New York Heart Association status, further cardiac catheterizations and operations, and current medications. Details of any catheterization or re-operation procedures were reviewed. In case the patient could not be contacted, their last known status was obtained from the referring cardiologist or primary care physician.

Statistics
Figures are reported as percentages of a common denominator (n = 28). Central tendency is reported as median and range unless otherwise specified. In view of the limited number of patients, a meaningful analysis for risk factors could not be undertaken. Survival analysis is depicted using the Kaplan-Meier method. Statistical calculations were performed using Statistica 6.0 (StatSoft Inc, Tulsa, OK).


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Patient Characteristics
There were 13 men (46%) and 15 women (54%). Seven patients (25%) were equal to or younger than 30 days (neonates), and 8 patients (29%) were between 30 and 365 days old (infants). The median age at operation was 7 months (range, 1 to 243).

Morphology
Ten patients (36%) were Lam type A, 17 patients (61%) were Lam subtype A1 and 5 patients (18%) were Lam subtype A2. Only 1 patient belonged in Lam type B. There was no patient with Lam type C in our series. The communication between the proximal and distal chamber was nonobstructive in 12 patients (43%). Fifteen patients (54%) had 33 associated congenital cardiac anomalies of which 20 (60%) were significant defects (Table 3). Nine patients (32%) had more than one other associated cardiac anomaly. Two patients (7%) had associated noncardiac anomalies (acrofacial dystosis [n = 1]; VATER (vertebral, anorectal, tracheal, esophageal, radius/renal anomalies) syndrome [n = 1]). Two patients (7%) had documented intrauterine growth retardation.


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  		Table 3. Associated Congenital Cardiac Anomalies
 
Presentation
Median age at presentation was 6 months (range, 0.6 to 240). Main presenting symptoms were as follows: tachypnoea (n = 22), failure to thrive (n = 12), poor feeding (n = 6), shock (n = 4), cyanosis (n = 3), decreased exercise tolerance (n = 2), exertional dyspnea (n = 2), respiratory arrest (n = 2), and increasing lethargy (n = 1).

Six patients (21%) with severe symptoms underwent emergency surgery (within 24 hours of admission). Another 8 patients (29%) were operated on during the initial hospital admission. Thirteen patients (46%) were operated on electively. Four patients (14%) were incidentally diagnosed. In 3 patients the opening in the diaphragm between the proximal and distal chamber was found to be restrictive, and the patients underwent surgical correction. One patient was incidentally diagnosed at 4 years of age. In view of the absence of symptoms and a nonrestrictive diaphragm, it was decided to only observe him regularly. Twenty-four patients (86%) had presented by 5 years, and all but 1 patient had presented by 10 years.

Diagnosis
Twenty-seven patients (96%) underwent a transthoracic echocardiography. Of these, 9 patients (32%), all with atypical cor triatriatum required cardiac catheterization for full demonstration of the defects. One patient underwent only cardiac catheterization, and 9 patients (32%) underwent both investigations. No patient underwent a transesophageal echocardiogram, which is probably a reflection of the era of our series. All patients with classic cor triatriatum (n = 13) were correctly diagnosed with preoperative transthoracic echocardiography. In all, 26 patients (96%) were correctly diagnosed before surgery. In 1 patient, the diagnosis was suspected and confirmed at operation.

Previous Operation
In 26 patients (93%) the surgical repair of cor triatriatum (and any associated cardiac anomaly) was the first cardiac operation. One patient had two previous modified Blalock-Taussig shunts. Two patients (7%) with atypical cor triatriatum had previous noncardiac surgery (repair of diaphragmatic hernia and imperforate anus [n = 1], repair of duodenal atresia [n = 1]).

Surgical Technique
Twenty-seven patients (96%) were treated surgically. All operations were performed through a median sternotomy. Cardiopulmonary bypass with systemic hypothermia and cardioplegic arrest was used in all patients. Deep hypothermic circulatory arrest was used in 8 patients (29%), 4 of whom had associated cardiac anomalies. The atrial approach was at the discretion of the surgeon. The defect was approached through the right atrium in 26 patients (93%). One 3-month-old female infant had the defect approached through the left atrium. The diaphragm between the proximal and distal chambers was completely excised taking care not to injure the mitral valve. The pulmonary venous ostia were all carefully identified at operation. The atrial septal defect (ASD) was repaired primarily or with a prosthetic or pericardial patch. The median cardiopulmonary bypass time was 55 minutes (range, 30 to 158). The median cross-clamp time was 29 minutes (range, 9 to 113), and the median deep hypothermic circulatory arrest time was 18 minutes (range, 8 to 42). Twenty-one patients (75%) underwent a total of 35 additional procedures (Table 4).


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  		Table 4. Additional Operative Procedures
 
Morbidity
Seventeen patients (60%) made an uncomplicated recovery. Ten patients (40%) had one or more complications. The main complications are shown in Table 5.


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  		Table 5. Morbidity
 
Early Mortality
There was 1 early death. A 3-month-old male infant with a diagnosis of atypical cor triatriatum (partial anomalous pulmonary venous drainage, large muscular ventricular septal defect, ASD) underwent an urgent operation to remove the left atrial membrane together with pulmonary artery banding and ASD closure. The initial recovery was uneventful. One week postoperatively, the interventional cardiologist attempted device closure of the ventricular septal defect. During cardiac catheterization the patient had complete heart block develop with profound hypotension. The procedure was abandoned. After the procedure the patient had seizures with signs of a significant cerebrovascular accident and complete absence of evoked sensory potentials. A computerized tomographic scan of the brain showed severe generalized ischemic cortical damage. Further treatment was discontinued.

Follow-Up
Four patients could not be located and were considered lost to follow-up. There was 1 early and 1 late death. Follow-up was 86% complete. The median follow-up interval was 98 months (range, 0.2 to 284). All patients including those with atypical cor triatriatum were in New York Heart Association functional class 1. One patient was taking warfarin for a Fontan circulation. No patient was taking any other medication at the time of follow-up. Eight preschool children (29%) had achieved normal developmental milestones for their age. Nine patients (32%) were full-time students and 8 patients (29%) had a regular job. One child with a preoperative diagnosis of cerebral palsy required full-time care.

Late Mortality
One patient died 10 years after repair. She presented with total anomalous pulmonary venous drainage, interrupted arch, pulmonary stenosis, ventricular septal defect, cor triatriatum, and dextrocardia. Prior to repair she had 2 previous modified Blalock-Taussig shunts. She underwent a complete repair at 20 years of age. She subsequently underwent an orthotopic heart transplant at 26 years of age and died 4 years later of cardiac causes. Kaplan-Meier survival was 96% (95% confidence interval, 75 to 99), 88% (95% confidence interval, 75 to 99) and 88% (95% confidence interval, 59 to 75) at 5, 10, and 15 years, respectively (Fig 1).



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  		Fig 1. Kaplan-Meier survival since operation. The number of patients available for follow-up at each 5-year interval is indicated.

 
Reoperation
One patient with atypical cor triatriatum involving mitral atresia, hypoplastic left ventricle, transposition of the great arteries, subaortic stenosis, and coarctation of the aorta underwent conversion to a total cavopulmonary connection (lateral tunnel Fontan) and a Damus-Kaye-Stanzel anastomosis 5 years after repair. Twelve years later he remains in the New York Heart Association functional class 1. No patient has required reoperation for pulmonary vein stenosis or residual or recurrent cor triatriatum.


   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
Cor triatriatum sinister can occur in isolation (classic) or in association with other congenital cardiac anomalies (atypical). Atypical cor triatriatum occurred in 54% of the patients in our series. A correct diagnosis was achieved in 27 patients (96%). One-third of our patients (all with atypical cor triatriatum) required cardiac catheterization for diagnosis. The presence of a communication between the proximal or distal chamber and the right atrium did not appear to reduce the severity of symptoms. Fifty-three percent of patients required treatment during infancy. There were no deaths immediately related to surgery for either classic or atypical cor triatriatum. Long-term results remain good with only 1 death 10 years after the repair.

The 3 main embryological theories explaining the development of cor triatriatum are malseptation, malincorporation, and entrapment [13–16]. Typically, the wall between the two chambers is thick and fibromuscular and has one or more openings in it. The foramen ovale is usually patent and may be in the septum between the right atrium and either the proximal or distal left atrial chamber. Sixty-one percent of patients in our series had an ASD (Lam type A1/A2) compared with 23% to 95% in earlier series (Table 1). Although there are other classification systems [17], the Lam classification has historically been used at our institute and also by others (Table 1). Anomalous pulmonary venous drainage (partial or total) is the most common associated abnormality; it has been reported in nearly one third of patients with cor triatriatum, which is similar to our series (29% patients) [5–10, 18]. A left superior vena cava coexists with cor triatriatum more frequently than with any other type of congenital heart disease (21% in our study) [18]. A wide variety of congenital cardiac anomalies including hypoplastic heart syndrome have been reported in association with cor triatriatum, although a review of published reports failed to discern the predominance of any single anomaly [19].

The natural history of cor triatriatum depends on the effective size of the opening in the diaphragm and the presence and location of the ASD [1, 18]. If the opening in the diaphragm is small and the ASD is absent or restrictive, the patient will present in infancy, and without treatment 75% of these patients will die [1, 18]. Neonates (< 30 days) and infants (< 1 year) accounted for 53% of patients [15] in our series. Eighty-six percent of patients (24 patients) had presented by 5 years of age, and all but 1 patient had presented by 10 years of age. Presentation can be delayed if there is a larger opening in the diaphragm and if the proximal pulmonary venous chamber communicates with the right atrium through an ASD permitting partial decompression. These patients often present later in childhood with signs and symptoms of mitral stenosis in the former and left-right shunting with resultant pulmonary over-circulation in the latter. Common symptoms then include tachypnoea, recurrent respiratory infections, and failure to thrive [20, 21]. However, in our series the presence of an ASD did not appear to reduce the severity of symptoms. Ten of 17 patients (59%) with an ASD required an urgent operation.

In patients with large communications the diagnosis may present only in adulthood. The proposed reasons for late conversion to a symptomatic state are believed to be caused by one or more of the following factors: fibrosis and calcification in the orifice of the separating diaphragm, the development of mitral regurgitation, and the development of atrial fibrillation [20]. Most cases of cor triatriatum presenting in adulthood are initially misdiagnosed as mitral valve stenosis [20, 21]. Atypical cor triatriatum usually presents early and the severity of symptoms is largely determined by the associated cardiac anomalies that often mask the presence of cor triatriatum [22]. In our series, 10 of 15 patients (66%) with atypical cor triatriatum presented in infancy, but 3 patients (20%) were older than 5 years of age at the time of presentation.

Earlier surgical series required cardiac catheterization to establish the diagnosis of cor triatriatum (Table 1) [7–9, 11]. Since the 1990s echocardiography has become the diagnostic modality of choice for classic cor triatriatum [23, 24]. We were able to achieve a correct preoperative diagnosis in 26 patients (96%). All patients with classic cor triatriatum and 6 of 15 patients (40%) with atypical cor triatriatum were correctly diagnosed using only transthoracic echocardiography preoperatively. Nine patients (32%) with atypical cor triatriatum did however require cardiac catheterization for diagnosis.

Treatment of cor triatriatum is primarily surgical. There are only two reports of successful balloon catheter dilation of the communication between the proximal and distal chambers [25, 26]. The follow-up periods for balloon dilatation were at 3 and 12 months, and the long-term outcome of balloon dilatation remains to be determined.

Patients with obstructive symptoms at any age should undergo surgical correction. The urgency of operation is primarily determined by the severity of the presenting symptoms. As cor triatriatum can remain asymptomatic until late adulthood, it is reasonable to observe patients with an incidental diagnosis, provided the opening in the diaphragm is nonrestrictive and regular follow-up is possible. We have had 1 such patient who was diagnosed at 4 years of age and who remains well and asymptomatic 18 years later. If adequate clinical follow-up is not possible, then surgical resection is probably justified.

The surgical approach can be through either a median sternotomy or a right thoracotomy. Most reported series have preferred the median sternotomy approach (Table 1) [7, 11]. We feel that a transseptal approach provides an excellent view while remaining technically easier in small children. The left atrial approach can be utilized in older children and adults with a dilated left atrium. A successful outcome depends on complete excision of the diaphragm between the proximal and distal chambers. The approach to atypical cor triatriatum is dictated by the nature of the associated anomalies.

Earlier surgical series have reported hospital mortality of 7% to 29% (Table 1). Most reported deaths occurred in children who presented in a critically ill condition, those with complex associated anomalies or after a misdiagnosis [5–11]. We have had no mortality related to surgery in our series in either the classic or atypical cor triatriatum. Reported long-term results of surgery are excellent and patient's life expectancy can be expected to approximate that of the general population [11].

In conclusion, cor triatriatum is a rare congenital cardiac anomaly. Surgical correction offers good early and long-term results for both classic and atypical cor triatriatum.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Brown J, Hanish SI. Cor triatriatum sinister, atresia of the common pulmonary vein, pulmonary vein stenosis, and cor triatriatum DexterIn: Mavroudis C, Backer C, editors. Pediatric Cardiac Surgery. Philadelphia, PA: Mosby; 2003. pp. 625-633.
  2. Church WS. Congenital malformations of heartabnormal septum in the left auricle. Trans Pathol Soc London 1868;19:188.
  3. Lewis FJ, Varco RL, Taufic M, Niazi SA. Direct vision repair of triatrial heart and total anomalous pulmonary venous drainage Surg Gynecol Obstet 1956;102:713.[Medline]
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