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Ann Thorac Surg 2009;88:440-444. doi:10.1016/j.athoracsur.2009.04.099
© 2009 The Society of Thoracic Surgeons
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Duccio Di Carlo
Roberto Di Donato
Bruno Murzi
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Giovanni Stellin
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Original Articles: General Thoracic

The Scimitar Syndrome: An Italian Multicenter Study

Vladimiro L. Vida, MD, PhDa, Simone Speggiorin, MDa, Massimo A. Padalino, MD, PhDa, Giancarlo Crupi, MDb, Carlo Marcelletti, MDc, Lucio Zannini, MDd, Alessandro Frigiola, MDe, Alessandro Varrica, MDe, Duccio Di Carlo, MDf, Roberto Di Donato, MDf, Bruno Murzi, MDg, Massimo Bernabei, MDg, Giovanna Boccuzzo, PhDh, Giovanni Stellin, MDa,*

a Pediatric and Congenital Cardiac Surgery Unit, University of Padua, Padua, Italy
b Ospedali Riuniti di Bergamo, Centro Diagnosi e Trattamento delle Cardiopatie Congenite, Bergamo, Italy
c Unità Operativa Cardiochirurgia Pediatrica, ARNAS Ospedale Civico e "Benfratelli," Palermo, Italy
d Cardiochirurgia Pediatrica, IRCS Gaslini, Genova, Italy
e Policlinico San Donato Milanese, Divisione di Cardiochirurgia, San Donato Milanese, Italy
f Ospedale Pediatrico Bambino Gesù, Servizio di Cardiochirurgia Pediatrica, Rome, Italy
g Unità Operativa di Cardiochirurgia Pediatrica, Ospedale "Pasquinucci," Massa, Italy
h Department of Statistics, University of Padua, Padua, Italy

Accepted for publication April 24, 2009.

* Address correspondence to Dr Stellin, Pediatric and Congenital Cardiac Surgery Unit, University of Padua, Via Giustiniani, 2, Padua, 35100, Italy (Email: giovanni.stellin{at}unipd.it).

Presented at the Poster Session of the Forty-fifth Annual Meeting of The Society of Thoracic Surgeons, San Francisco, CA, Jan 26–28, 2009.


   Abstract
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 Abstract
Introduction
 Material and Methods
 Results
 Comment
 References
 
Background: Scimitar syndrome is a rare congenital heart disease. To evaluate the results of managing this malformation surgically, we have embarked on a multicenter Italian study involving seven different centers and reporting the largest published series in the medical literature.

Methods: From January 1997 to December 2007, 26 patients with scimitar syndrome who underwent surgical correction were included. Primary outcomes include hospital mortality and the efficacy of repair at the follow-up.

Results: Median age was 11 years (interquartile range, 1.8 to 19.9 years). Nineteen patients (73%) presented with symptoms including upper respiratory tract infections (n = 13), recurrent pneumonia (n = 10), cardiac failure (n = 4), and cyanosis (n = 2). Associated cardiac anomalies were present in 16 patients (63%). Surgical repair included intraatrial baffle repair in 18 patients (69%; group 1), and reimplantation of the "scimitar vein" onto the left atrium in 8 patients (31%; group 2). One patient died in hospital (3.8%; group 1). Postoperative complications were less frequently reported in group 1 (4 of 18 patients, 22%) compared with group 2 (5 of 8 patients, 62%). Median follow-up time was 4 years (interquartile range, 1.8 to 9.7 years). There was 1 late death (1/25 patients, 4%; group 2). Four patients (16%) showed a complete occlusion of the scimitar drainage (2 in group 1, 12%; 2 in group 2, 25%) and 3 patients (12%) required balloon dilation or stenting for scimitar vein stenosis (1 in group 1, 6%; 2 in group 2, 25%).

Conclusions: The intraatrial baffle repair seems to have a lower incidence of postoperative complications and a better patency rate, at last follow-up, than the reimplantation of the scimitar vein onto the left atrium.


   Introduction
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 Abstract
 Introduction
Material and Methods
 Results
 Comment
 References
 
Scimitar syndrome is a rare association of congenital cardiopulmonary anomalies consisting of a partial or total anomalous pulmonary venous connection of the right lung to the inferior vena cava, right lung hypoplasia, and systemic arterial supply of a variable degree to the right lung [1–6]. This syndrome can present early in the neonatal period or later in life, with a wide clinical spectrum [7–12].

Surgical correction of this malformation is usually accomplished in symptomatic patients or in patients with an increased pulmonary blood flow and signs of right heart chamber dilatation [5–8]. Two different surgical approaches have been developed and used, according to the surgeon's preference and to the anatomic and pathologic features of each case [13–22]. So far, there is no consensus in the literature on the best option for surgical treatment.

The object of this study is to analyze the early surgical results and the follow-up outcome of patients who underwent surgery for scimitar syndrome within a multicenter Italian study data collection, which is the largest experience published to date.


   Material and Methods
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 Abstract
 Introduction
 Material and Methods
Results
 Comment
 References
 
A review of the medical records and computerized hospital data was approved by the Clinical Investigation Committee from the University Hospital of Padua, and the procedures followed were in accordance to the institutional guidelines for retrospective record review and protection of patient confidentiality. Individual consent was not obtained for patients enrolled in this study. Patients are not identified, and the chairperson of the ethics committee of each institution consented to send their data for publication.

The data reviewed was relative to the hospital course, and to the follow-up clinical controls of patients with scimitar syndrome who required surgical treatment between January 1997 and December 2007. Seven Italian cardiothoracic centers contributed to the data collection. Excluded were 2 patients who were treated with a right lung pneumectomy. These last 2 patients, 59 and 81 days old, had severe lung hypoplasia and presented with congestive heart failure. One of them also had severe recurrent respiratory tract infections.

Predictor variables include demographic and preoperative clinical data, different operative techniques, and follow-up variables, including clinical and instrumental information.

Primary outcomes include the hospital mortality and the patency of the scimitar vein at the last control. Follow-up outcomes were evaluated only in patients who had surgical repair, including intraatrial baffle technique or direct scimitar vein reimplantation. Excluded were patients who were treated with a right lung pneumonectomy.

Quantitative variables were summarized as median and interquartile range, equal to the difference between the first (25% of the distribution) and the third (75% of the distribution) quartiles. Because quantitative variables are not normally distributed, comparison among groups was carried out by the Wilcoxon signed-rank test [23]. Difference between proportions was assessed by means of the Fisher's exact test [24].

Data were analyzed using SAS software, release 9.1.3 (SAS Institute Inc. SAS Stat 9.1, Cary, North Carolina) [25], and probability values less than 0.05 were considered significant.


   Results
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 Abstract
 Introduction
 Material and Methods
 Results
Comment
 References
 
Twenty-six consecutive patients who underwent surgical treatment for scimitar syndrome in 7 different Italian centers were included in this study. There were 16 female patients (61%) and 10 males (39%). Preoperative diagnosis was achieved by echocardiography in all of them. Additional instrumental examinations included cardiac catheterization in 22 patients (85%) and magnetic resonance in 3 patients (11%). Nineteen patients (73%) presented with symptoms, including upper respiratory tract infections in 13 patients (50%), recurrent pneumonia in 10 patients (38%), cardiac failure in 4 patients (15%), cyanosis in 2 patients (7.6%), supraventricular tachycardia in 2 patients (7.6%), and growth retardation in 1 patient (3.8%). Associated cardiac anomalies included atrial septal defect type II in 16 patients (63%), dextrocardia in 7 patients (27%), right lung hypoplasia in 7 patients (27%), total anomalous pulmonary venous return in 1 patient (3.8%), ventricular septal defect in 1 patient (3.8%), and severe mitral valve regurgitation in 1 patient (3.8%).

At preoperative cardiac catheterization, the median systolic pulmonary artery pressure was 25.5 mm Hg (interquartile range, 21.5 to 30 mm Hg) and the preoperative median pulmonary blood flow to systemic blood flow ratio was 2:1 (interquartile range, 2:1 to 2.5:1). Systemic arterial supply to the right lung was demonstrated in 4 patients (15%); 4 of them were treated by coil embolization of aortopulmonary collaterals at the time of preoperative study. Pulmonary sequestration was seen in 2 patients (7.7%).

Median age at surgery was 11 years (interquartile range, 1.8 to 19.9 years). Four patients (15%) were less than 12 months of age and 10 patients older than 16 years (38%). Age distribution among patients with and without complications is the same by Wilcoxon signed-rank test (p = 0.46). Surgical approach was through a midline sternotomy in 25 patients (96%) or through a right thoracotomy in 1 patient (4%).

Surgical repair included group 1 with 18 patients (69%) undergoing an intraatrial baffle repair; whereas group 2 had 8 patients (31%) undergoing a reimplantation of the scimitar vein on the left atrium. Associated surgical maneuvers in 17 patients (65%) were atrial septal defect type II closure in 16 patients, total anomalous pulmonary venous connection repair in 1 patient, ventricular septal defect closure in 1 patient, mitral valve replacement in 1 patient, and tricuspid valve plasty in 1 patient. Fourteen patients (54%) were treated by hypothermic core cooling and circulatory arrest (Table 1). Two patients had neurologic events, both of them after circulatory arrest.


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  		Table 1 Patient Characteristics and Outcomes According to Corrective Surgical Technique a
 
Postoperative complications presented in 9 patients (35%; Table 2) and are equally distributed by age. One patient in group 2 was reoperated on during the same hospitalization for stenosis at the inferior vena cava level.


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  		Table 2 Postoperative Complications (13 Complications in 9 Patients)
 
Median intensive care unit time and hospitalization time are reported in Table 1. Postoperative complications were less frequently reported in group 1 (4 of 18 patients, 22%) compared with group 2 (5 of 8 patients, 62%; Tables 1, 2).

One patient died in hospital (3.8%) as a result of severe brain injury, 56 days after an initial intraatrial baffle repair (Table 1).

Median follow-up time was 4 years (interquartile range, 1.8 to 9.7 years) and was completed in all.

One patient died late (4%), 126 days after scimitar vein reimplantation onto the left atrium, ligation of a patent ductus arteriosus, and a ventricular septal defect closure. Cause of death was severe pulmonary arterial hypertension causing repeated pulmonary hypertensive crises, leading to an eventual cardiac arrest. Lung biopsy obtained at the time of surgical repair showed Heath-Edwards class III pulmonary vascular obstructive disease.

At the last clinical follow-up control, 22 of 24 patients (92%) are in New York Heart Association functional class I. Among the remainder, 1 presented with atrial fibrillation, 1 with recurrent respiratory infection, and 1 with cyanosis.

At the echocardiographic or cineangiographic control studies, 7 patients (29%) showed either a stenosis or an occlusion of the scimitar vein. There were 1 stenosis and 2 occlusions in group 1 (18%) and 2 stenoses and 2 occlusions in group 2 (50%; Fig 1). None of them presented hemoptysis.


Figure 1
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  		Fig 1. Freedom from scimitar vein obstruction or stenosis at follow-up (according to corrective surgical technique). (Solid line = intraatrial baffle; dashed line = direct reimplantation.)

 
Balloon dilatation and stenting was performed in 3 patients (1 patient in group 1, 6%; 2 patients in group 2, 25%), 8, 12, and 15 months after correction, respectively. None of the patients underwent reoperation.

At multivariate analysis (logistic regression) no variables were significantly associated with outcomes.


   Comment
Top
 Abstract
 Introduction
 Material and Methods
 Results
 Comment
References
 
Scimitar syndrome is a rare congenital heart disease characterized by a wide spectrum of lesions linked to the anomalous right pulmonary venous drainage and to the severity of right lung hypoplasia [1–4, 26]. The triad of respiratory distress, right lung hypoplasia, and dextroposition of the heart should alert the clinician to the possibility of this syndrome [5, 6]. In addition the plain chest x-ray film is a very relevant tool in the screening of this disease, because it discloses a persisting right paracardiac image, as a result of abnormal right pulmonary venous drainage to inferior vena cava ("scimitar sign") [27].

Because of the wide range of symptoms at clinical onset, the diagnosis may be difficult, especially in children and young adults with concomitant congenital heart lesions [1–4, 28–31].

Anomalous systemic arterial supply, pulmonary vein stenosis, and associated cardiovascular anomalies play a significant role in these patients, and the ultimate outcome of individual infants depends on the feasibility of treating these anomalies, especially in early infancy [7–9]. Moreover, especially in patients with associated other congenital heart diseases, a delayed surgical treatment can lead to pulmonary vascular obstructive disease, which can jeopardize the surgical outcome [10].

Currently, there is no consensus regarding the best surgical treatment option for these patients. Many surgical techniques have been proposed; however, the current literature reports only a few studies with limited comprehensive experience on the subject [13–22].

Simple ligation or coil embolization of the abnormal arterial vessels has been advocated as the best and simplest type of treatment, especially in symptomatic infants, often improving clinical symptoms [7–13]. Huddleston [8], on the other hand, reported that in symptomatic patients, although repair of the anomalous venous return and ligation of collaterals is generally recommended, right pneumonectomy (either as primary therapy or if repair failed) showed similar early and late results.

In our multicenter Italian study we are reporting the largest experience so far published in the medical literature with the aim of analyzing the type of repair performed, the surgical risk, and the midterm results at follow-up. Our data are indicating that scimitar syndrome is not a simple and benign congenital heart disease, and it is often associated with other cardiac and extracardiac anomalies, which play an important role for the long term. The majority of patients presented with preoperative respiratory symptoms, as upper respiratory infections and recurrent pneumonia episodes. These symptoms may be confusing at the beginning, together with the fact that anomalous presentation of the syndrome can occur [1, 4, 10, 32]. However, after the diagnosis has been correctly established, the presence of respiratory symptoms usually represents one of the main indications for surgical correction. Furthermore, surgical repair seldom results in normal blood flow to the right lung, abolishing the increased left-to-right shunt with pulmonary volume overload that often results in improving clinical conditions in the majority of patients [10].

According to our data, surgical correction can be done safely with a low mortality rate, independent of these two surgical techniques. The direct reimplantation technique, however, carries a higher incidence of postoperative complications that may be related to the longer hospital stay in this group of patients.

The main problem, however, was that these patients were represented by the exceptionally high incidence of postoperative pulmonary venous obstruction and abnormally diminished perfusion of the right lung [2, 10]. Our overall absence of scimitar vein obstruction or stenosis was 71% at the last follow-up check, and this was higher for patients who underwent an intracardiac baffle repair compared with those who had a reimplantation of the scimitar vein (82% versus 50%). It is of note that scimitar vein obstruction or stenosis is not age-related and the majority of the patients presented good clinical conditions at the last clinical control. A limitation of this study is represented by the fact that in only a few patients the right lung performance was assessed by pulmonary scintigraphy, which could have contributed to giving more detailed information about right lung function and therefore the patient's clinical status.

A consideration should also be made of the fact that follow-up studies of nonoperatively managed patients with scimitar syndrome showed that they can lead a normal life, but therapeutic approach for patients with respiratory manifestations and onsets beyond the neonatal period should be conservative [32]. An awareness of this syndrome may avoid unnecessary invasive diagnostic procedures, surgical treatment, and subsequent need for interventional catheter procedures for most patients [12, 13] without compromising symptoms. Magnetic resonance imaging is now playing an important role in diagnosing and monitoring the cardiopulmonary anatomy and particularly the right lung structure in these patients [33–37].

In conclusion, although our data are too small for drawing a statistical conclusion, it seems that the intracardiac baffle technique carries a lower incidence of postoperative complications and a better patency rate in the midterm.

A larger multicenter experience and longer follow-up are necessary for drawing a definitive conclusion about the best treatment option for this rare pathologic process.


   References
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 Abstract
 Introduction
 Material and Methods
 Results
 Comment
 References
 

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