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J Thorac Cardiovasc Surg 1994;107:1536-1538
© 1994 Mosby, Inc.

LETTERS TO THE EDITOR

Complete atrioventricular canal and total anomalous pulmonary venous drainage: A rare association

A. D. Abbattista , MD*Go, B. Marino , MD, F. S. Iorio , MD, C. Marcelletti , MD

Pediatric Hospital Bambino Gesù
Department of Pediatric Cardiology and Cardiac Surgery
Rome, Italy

To the Editor:

Atrioventricular canal (AVC) and total anomalous pulmonary venous drainage (TAPVD), apart from cases with atrial isomerism, is such a rare association of congenital cardiac malformation that, to our knowledge, only four cases have been reported so farGo Go 1,2

A 6-month-old female infant (weight 6.5 kg) was sent to our institution for congestive heart failure. On admission, two-dimensional Doppler echocardiography showed a complete AVC-Rastelli type A, with a single papillary muscle of the left ventricle and a supracardiac form of TAPVD. To visualize the exact site of the pulmonary venous connection, we performed an angiographic study. Left pulmonary veins drained through a vertical vein into the superior vena cava, and right pulmonary venous return was found to have a double connection,Go 3 draining partly into a stenotic vertical vein and partly into the coronary sinus through a single venous channel (Figs. 1 and 2).



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  		Fig. 1. Angiographic injection in left pulmonary artery shows left pulmonary veins draining to right superior vena cava through vertical vein.

 


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  		Fig. 2. Angiographic injection in left pulmonary artery shows right pulmonary veins draining to coronary sinus (black arrow) and to vertical vein (white arrow).

 
The patient underwent surgical correction at the age of 8 months. The operation was performed with deep hypothermic circulatory arrest. The right atrium was incised to the base of left auricular appendage and anastomosed with the venous channel, which had been lanced in the same way. The right edge of the venous channel was reapproached and the posterior wall of the right and left atria enlarged with a polytetrafluorocthylene patch. The connection between the venous channel and coronary sinus was interrupted, and the AVC was repaired with a double patch technique. The mitral cleft was partially sutured because a single papillary muscle was present. St. Thomas' Hospital crystalloid cardioplegic solution was administered before and after 60 minutes of circulatory arrest. Sternal closure was delayed for 3 days, and the postoperative period was complicated by rhythm disturbances and the need for external pacing. Sinus rhythm was regained after 1 week and the baby was discharged 3 weeks later.

For isolated TAPVD, two-dimensional Doppler echocardiography alone has been suggested.Go 4 We report this rare association of AVC and TAPVD to suggest a preoperative angiocardiographic study when TAPVD is accompanied by other anomalies or when a mixed type or a double connection is suspected. Once more, the presence of AVC in patients without Down syndrome is linked with left-sided obstruction.Go Go Go 1,5,6 In this case, the form was obstructed pulmonary venous return with double connection and single papillary muscle of the left ventricle. Last, the good operative and follow-up results demonstrate the feasibility of optimal surgical correction when a clear preoperative view is obtained.

We thank G. Salvatori and Dr. B. Sebastiani of National Research Council for their kind collaboration.

References

  1. Penkoske PA, Neches WH, Anderson RH, Zuberbhuler JR. Further observations on the morphology of atrioventricular septal defects. J THORAC CARDIOVASC SURG 1985;90:611-22. [Abstract]
  2. Yee ES, Turley K, Hsieh WS, Ebert PA. Infant total anomalous venous connection: factors influencing timing of presentation and operative outcome. Circulation. 1987;76(Suppl):III83.
  3. Arciprete P, McKay R, Watson GH, Hamilton DI, Wilkinson JL, Arnold RM. Double connections in total anomalous pulmonary venous connection. J THORAC CARDIOVASC SURG 1985;92:145-52.
  4. Hutha JC, Gutsegell HP, Nihill MR. Cross sectional echocardiographic diagnosis of total anomalous pulmonary venous connection. Br Heart J 1985;53:525-34. [Abstract/Free Full Text]
  5. Starc TJ, Bierman FZ, Bowman FO, Steeg CN, Wang NK, Krongrad E. Pulmonary venous obstruction and atrioventricular canal anomalies: role of cor triatriatum and double outlet right atrium. J Am Coll Cardiol 1987;9:830-3. [Abstract]
  6. Marino B, Vairo U, Corno A, Marcelletti C. Atrioventricular canal in Down syndrome. Am J Dis Child 1990;144:1120-2.[Abstract/Free Full Text]



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Home page
 Eur. J. Cardiothorac. Surg.Home page
V. Alexi-Meskishvili, I. Dahnert, E. Beyer, and R. Hetzer
Successful total correction of complete atrioventricular canal, total anomalous pulmonary venous drainage and unroofed coronary sinus in an infant
Eur. J. Cardiothorac. Surg., January 1, 1999; 15(1): 95 - 96.
[Abstract] [Full Text] [PDF]

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