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Authors:




Department of Pediatric Cardiology, Escorts Heart Institute and Research Center (EHIRC), Okhla road, New Delhi,110025,India.


Correspondence to: Dr Department of Pediatric Cardiology, Escorts Heart Institute and Research Center (EHIRC), Okhla road, New Delhi,110025,India


Scimitar Syndrome: A novel management approach for palliation in a sick infant





Authors:




Department of Pediatric Cardiology, Escorts Heart Institute and Research Center (EHIRC), Okhla road, New Delhi,110025,India.


Correspondence to: Dr Department of Pediatric Cardiology, Escorts Heart Institute and Research Center (EHIRC), Okhla road, New Delhi,110025,India







ABSTRACT
Scimitar Syndrome: A novel management approach for palliation in a sick infant

Scimitar syndrome is a rare congenital anomaly with essentially surgical management. Nonsurgical interventional treatments are rarely possible but is associated with poor outcome. We report a case of Schmitar syndrome with obstructed partial pulmonary venous confluence, large PDA, severe PAH where significant decrease in pulmonary artery pressures was achieved by pulmonary venous stenting in combination with coiling of aberrant vascular supply to the sequestered lung statement. To the best of our knowledge no such procedure in combination has not been reported in the literature.
 
Key Words : Scimitar syndrome, Pulmonary venous stenting








Scimitar Syndrome: A novel management approach for palliation in a sick infant

Scimitar syndrome is a rare congenital anomaly accounting for about 0.5% of all congenital heart diseases with poor outcome in infantile age group. It is characterized by anomalous drainage of  right side pulmonary veins,lung sequestration with aberrant vascular supply and lung hypoplasia. Nonsurgical treatments are rarely possible  and are associated with poor outcome We report a rare case where significant decrease in pulmonary artery pressures was achieved by interventional treatment, by stenting of the obstructed pulmonary venous confluence in combination with coiling of aberrant vascular supply to the sequestered lung segment followed by ductal ligation.Child showed maked improvement thereafter and was discharged in a stable condition on home oxygen therapy. 

CASE REPORT

Baby G, 6-month old female infant was first in birth order. She was product of full term cesarean delivery with a birth weight of 2.5 kg. Child became symptomatic at 4 months of age in the form of respiratory distress. She was initially evaluated for chest infection and was further referred as suspected heart disease to our center. Child was tachypnic and in congestive heart failure at presentation. she had a saturation of 65%. Echo evaluation showed partial anomalous pulmonary venous drainage (PAPVC) of the right upper and lower pulmonary veins drainage into inferior vena cava (IVC) with obstruction. at its site on confluence with IVC with mean peak gradient of 5 mm hg and loss of phasic flow  Fossa ovalis ASD, 4mm with right to left shunt, PDA (3.8 mm) with bi-directional shunting and severe pulmonary artery hypertension (PAH). Child was further evaluated by a CT angiography scan which showed localized stenosis of the right pulmonary venous confluence to IVC with right lung hypoplasia and sequestration of the right posterobasal segment having separate supply from Aorta. Child continued to have features of congestive heart failure inspite of optimization of the decongestive therapy and was thus undertaken for the interventional procedure. 
 She underwent stenting of the pulmonary venous confluence to IVC with balloon mounted Genesis stent. The supply to the sequestered lung segment from the descending aorta below diaphragm was coiled with a Cook coil 0.038x4x4. Child was electively ventilated with very slow and gradual weaning of ventilatory settings in the post interventional period. She showed fall in the pulmonary artery pressure with peak PG of 48 mmhg (against systemic pressures of 80 mm hg) although pulmonary artery pressures continued to be on a higher side making it difficult to wean from the ventilator. Echo showed left to right shunt. In view of the symptomatic status with large PDA, child was undertaken for PDA ligation. Child had a smooth postoperative course, was weaned from the ventilator over a period of 12 days. She was discharged on home-based oxygen therapy basal desatuartion off oxygen. However a month later child developed fever with sudden onset respiratory distress and died from respiratory tract infection.




DISCUSSION
Scimitar syndrome (SS) is a rare association of congenital cardiopulmonary anomalies with  incidence of approximately 1 to 3 per 100,000 live births[1],consisting of a partial anomalous pulmonary venous connection of the right lung to the inferior vena cava, right lung hypoplasia, dextroposition of the heart, and anomalous systemic arterial supply to the right lung. First described in 1836 term � Schmitar� was coined because of the radiographic appearance of the anomalous vein, resembling a curved Turkish sword or Scimitar.[2].  Intervention therapy is rarely described for this condition in the English literature.
Infantile Schmitar syndrome unlike adult form becomes symptomatic soon after birth. as in our case Their course is often complicated by severe PAH and cardiac failure, Pulmonary hypertension is often recognized as the cause of the severe symptoms and poor outcome of SS during infancy.[3.4] Multiple factors responsible for PAH include :large left-to-right shunt via the anomalous pulmonary vein, left-to-right shunt from the systemic arterial supply to the right lung, right lung hypoplasia with reduction of the pulmonary vascular bed ,pulmonary vein stenosis and obstruction, other associated congenital cardiac malformations, persistent pulmonary hypertension This was well seen in our case as after the interventional procedure child continued to have PAH because of the large shunt (left to right ) through PDA .Pulmonary venous stenosis is a well known cause of PAH in infants with Scimitar syndrome and was well present in our case too. An attempt to relieve the venous obstruction by pulmonary venous stenting has not been reported in the English literature in such cases. Interventional procedures in the literature primarily involve coiling or embolization of the arterial supply to the sequestered lung segment. We undertook coiling of the sequestered lung segment and decided to undertake pulmonary venous stenting in view of the sick child unsuitable for surgical intervention.We understood the fact that this will itself contribute to increased left to right pretricuspid shunting but was essential as xary features showed marked pulmonary venous obstructive changes in right lung field. The pulmonary pressures dropped significantly after stenting evident by the left to right shunting of the PDA . Child continued to have features of CHF making him difficult to wean off the ventilator, thus she was taken for off bypass PDA ligation. 
 It may be difficult to determine which of these many factors are responsible for the pulmonary hypertension in an individual patient, so each factor should be considered and investigated. Multiple factors as in our case might be simultaneously operative and need to be addressed individually. management requires both surgical and interventional procedures, besides a good supportive care. One center reported the results of treatment of 56 symptomatic infants in a compilation of 3 large series of patients.[5] The patients received a variety of medical and surgical treatments with an overall mortality of 45%.6,11 Early intervention may be warranted as in our case to control high pulmonary artery pressures. Intervention procedures such as stenting of the obstructed pulmonary vein may be done as a emergent palliative treatment to relieve PAH with good results . These may need to be further supported by surgical assistance to decrease the pulmonary pressures like PDA ligation as in our case.
       


                                            References


2. Neill CA, Ferencz C, Sabiston DC, Sheldon H. The familial occurrence of hypoplastic right lung with systemic arterial supply and venous drainage "scimitar syndrome." Bull Johns Hopkins Hosp. 1960;107:1-15. 
1. Dupuis C, Charaf LAC, Breviere GM, Abou P, Remy-Jardin M, et al. The "adult" form of the scimitar syndrome. Am J Cardiol. 1992;70:502-507.
 
5Canter CE, Martin TC, Spray TL, Weldon CS, Strauss AW. Scimitar syndrome in childhood. Am J Cardiol. 1986;58:652-654. 
3.Huddleston CB, Exil V, Canter CE, Mendeloff EN. Scimitar syndrome presenting in infancy. Ann Thorac Surg. 1999;67:154-160. 
 
4Mardini MK, Sakati NA, Lewall DB, Christie R, Nyhan WL. Scimitar syndrome. Clin Pediatr. 1982;21:350-354. 
 
6.Inoue T, Ichihara M, Uchida T, Sakai Y, Hayashi T, et al. Three-dimensional computed tomography showing partial anomalous pulmonary venous connection complicated by the scimitar syndrome. Circulation. 2002;105:663. 



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