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Asian Cardiovasc Thorac Ann 2002;10:262-263
© 2002 Asia Publishing EXchange Pte Ltd

CASE STUDY

Congenital Cleft of Anterior Tricuspid Leaflet in Adolescent

Hüseyin Okutan, MD, Turhan Yavuz, MD, Selçuk Bilgin, MD1, Harun Düver, MD, Ali Kutsal, MD

Department of Thoracic and Cardiovascular Surgery
1 Department of Anesthesiology Sevket Demirel Heart Center Süleyman Demirel University Medical School Isparta, Turkey
For reprint information contact: Hüseyin Okutan, MD Tel: 90 246 232 6657 Fax: 90 246 232 4510 email: okutanh{at}yahoo.com Department of Thoracic and Cardiovascular Surgery, Sevket Demirel Heart Center, Süleyman Demirel University Medical School, Isparta 32100, Turkey.

   ABSTRACT
TOP
 ABSTRACT
INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 
The diagnostic findings and treatment of an isolated congenital cleft of the anterior leaflet of the tricuspid valve in a 14-year-old boy are described. An atrial septal defect was closed by primary suturing, and the tricuspid valve was successfully reconstructed by De Vega annuloplasty.


   INTRODUCTION
TOP
 ABSTRACT
 INTRODUCTION
CASE REPORT
 DISCUSSION
 REFERENCES
 
Severe primary tricuspid regurgitation (TR) in an adolescent is unusual. Congenital tricuspid insufficiency due to a cleft in the anterior tricuspid leaflet is a rare congenital anomaly.1–3 Most reported cases were associated with a perimembranous ventricular septal defect, pulmonary stenosis, or atrial septal defect (ASD).


   CASE REPORT
TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
DISCUSSION
 REFERENCES
 
A 14-year-old boy referred for repair of an ASD was in New York Heart Association functional class II with exertional fatigue as the limiting symptom. A holosystolic murmur was noted at the mid-cardiac region. Laboratory tests and electrocardiography were normal. Chest radiography findings were nonspecific. Echocardiography showed an ASD (secundum type), pulmonary hypertension (45 mm Hg), TR (2 to 3 degrees), mitral regurgitation (1 to 2 degrees), and Qp/Qs of 1.9 (Figure 1Go). Using bicaval and aortic cannulation, continuous cardio-pulmonary bypass at 28°C, and crystalloid cardioplegia, a right atriotomy was performed. A cleft of the anterior tricuspid leaflet was found to be associated with an ostium secundum ASD (Figure 2Go). The tricuspid annulus was 35 mm in diameter. There was no abnormality in the chordal attachment. The cleft ran through the anterior leaflet to the annulus, it was repaired with 3 interrupted 6/0 polypropylene sutures. The ASD was closed by primary suturing, and the tricuspid valve (TV) was reconstructed by De Vega annuloplasty (Figure 3Go). The patient recovered uneventfully. Echocardiography before discharge demonstrated grade 1 mitral incompetence and minimal incompetence of the TV. Five months postoperatively, his clinical status was excellent with no echocardiographic signs of tricuspid incompetence.



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  		Figure 1. Preoperative echocardiography showing an ostium secundum ASD. ASD = atrial septal defect, LA = left atrium, LV = left ventricle, RA = right atrium, RV = right ventricle.

 


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  		Figure 2. Schematic representation of the cardiac anomalies. ASD = atrial septal defect.

 


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  		Figure 3. Diagram showing the repaired atrial septal defect and reconstructed tricuspid valve.

 

   DISCUSSION
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 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
REFERENCES
 
The diagnosis of TR (classified as primary, secondary, or physiologic) can be established by 2-dimensional echocardiography.4 Right ventricular volume or pressure overload can cause secondary TR with normal valve anatomy. Causes of TR include Ebstein’s anomaly, TV prolapse, rheumatic valve disease, endocarditis, trauma, dysplasia, or dysfunction after right ventricular infarction.5 A cleft of the TV is a rare Doppler echocardiographic finding.1 In adolescents and adults examined routinely by Doppler echocardiography, the incidence of cleft was 1:161 patients (0.6%).6 In another study, this anomaly was found in 5 of 28,091 Doppler echocardiograms (0.018%).1 However, the cleft in this patient could not be demonstrated by Doppler echocardiography.

In previous cases, the reason for TR was dysplasia of the TV.1 In contrast, clefts of the mitral valve are well known and associated with atrioventricular canal defects.1 Eichhorn and colleagues1 postulated two explanations for the occurrence of a cleft: in all of their cases, the cleft was located in the anterior leaflet close to where the lateral endocardial cushion meets the right dorsal conus swelling, thus the cleft might be the result of anomalous fusion; another possible explanation is the great variability in anatomic structure of the TV. The treatment of this rare cardiac anomaly is surgical reconstruction.


   REFERENCES
TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 REFERENCES
 

  1. Eichhorn P, Ritter M, Suetsch G, von Segesser LK, Turina M, Jenni R. Congenital cleft of the anterior tricuspid leaflet with severe tricuspid regurgitation in adults. J Am Coll Cardiol 1992;20:1175–9.[Abstract]

  2. Tatebe S, Miyamura H, Watanabe H, Sugawara M, Eguchi S. Closure of isolated ventricular septal defect with detachment of the tricuspid valve. J Card Surg 1995; 10:564–8.[Medline]

  3. Ogus NT, Naseri E, Arsan S. Congenital tricuspid insufficiency due to a cleft in tricuspid anterior leaflet associated with perimembranous VSD. An unusual case report. Turk J Pediatr 1998;40:627–8.[Medline]

  4. Choong CY, Abascal VM, Weyman J, Levine RA, Gentile F, Thomas JD, et al. Prevalence of valvular regurgitation by Doppler echocardiography in patients with structurally normal hearts by two-dimensional echocardiography. Am Heart J 1989;117:636–42.[Medline]

  5. Waller BF, Moriarty AT, Eble JN, Davey DM, Hawley DA, Pless JE. Etiology of pure tricuspid regurgitation based on anular circumference and leaflet area: analysis of 45 necropsy patients with clinical and morphologic evidence of pure tricuspid regurgitation. J Am Coll Cardiol 1986;7:1063–74.[Abstract]

  6. Eichhorn P, Sutsch G, Jenni R. Congenital heart defects and abnormalities newly detected with echocardiography in adolescents and adults [German]. Schweiz Med Wochenschr 1990;120:1697–700.[Medline]





This Article
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