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Year : 2004  |  Volume : 14  |  Issue : 4  |  Page : 405-407
Pulmonary artery sling - a case report


Head of Department of CT, MRI and DSA Jaslok Hospital and Research Center Mumbai, India

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Keywords: Pulmonary Artery Sling, Anomalous pulmonary arterial supply, CT

How to cite this article:
Hedge A N, Halankar J A, Desai S B. Pulmonary artery sling - a case report. Indian J Radiol Imaging 2004;14:405-7

How to cite this URL:
Hedge A N, Halankar J A, Desai S B. Pulmonary artery sling - a case report. Indian J Radiol Imaging [serial online] 2004 [cited 2019 Sep 16];14:405-7. Available from: http://www.ijri.org/text.asp?2004/14/4/405/28682

   Introduction Top


Pulmonary Artery sling is a rare condition resulting from an anomalous origin of the left pulmonary artery from a normally positioned right pulmonary artery. Glaevecke and Doehle first described it in 1897[1]. It usually occurs in neonates and young infants, is often associated with tracheobronchial and cardiovascular anomalies, and is almost always fatal if left untreated. [2]

We report a case of Pulmonary Artery sling detected in a neonate on a Multislice CT pulmonary angiogram. Also incidentally noted was an anomalous artery arising from the abdominal aorta, just superior to coeliac artery and was seen to supply the medial basal segment of the right lower lobe.


   Case Top


A 19-day-old female child, product of a non-consanguineous marriage was referred with the complaints of severe respiratory distress, expiratory wheeze and cyanosis. An echocardiogram done at another center had demonstrated an Atrial Septal Defect, enlarged main pulmonary artery and right cardiac chambers with a mild Tricuspid regurgitation. Left pulmonary artery was not visualized on echocardiography. The child was advised to undergo a CT pulmonary angiography for a detailed evaluation of the pulmonary arteries.

The CT Pulmonary angiogram was obtained on a 16 slice GE Light speed MDCT machine after administering 30 cc of non ionic contrast (concentration 400 mg%) by hand injection at a rate of 1cc per second. Images were obtained in the caudo cranial direction. The main pulmonary artery, right atrium and right ventricle were enlarged. The right pulmonary artery was normally positioned.

The left pulmonary artery however, was seen to branch out from the right pulmonary artery and was seen to curve around the trachea, just above the carina. It was then seen to traverse between the trachea and the esophagus, reach the left hilum and then branch into its smaller regular divisions. The sling compressed the trachea. Also incidentally noted was an anomalous artery arising from the abdominal aorta just superior to origin of the coeliac artery and it was seen to traverse superiorly to supply the medial basal segment of the right lower lobe. However, the pulmonary parenchyma in the right lower lobe was normal.


   Discussion Top


Pulmonary artery sling is an anomalous origin of the left pulmonary artery from the right pulmonary artery. It then courses posteriorly over the right main bronchus near its origin from the trachea, traverses between the trachea and the esophagus and enters the left hilum. Although it is most frequently diagnosed in symptomatic children, the condition can be found in asymptomatic adults in whom it can mimic a mediastinal adenopathy. [3].

This probably occurs due to the involution of the proximal left sixth arch [4]. An anastomotic vessel linking the primitive pulmonary circulations becomes the anomalous left pulmonary artery, which arises, from the right pulmonary artery and courses between trachea and esophagus.

Incidence of associated congenital malformations varies and has been noted to occur in 81% in one study [5] and primarily involve the tracheobronchial tree and cardiovascular system.

Tracheobronchial anomalies include a bronchus arising directly from the trachea and supplying a segment of the right lung, hypoplasia of the distal trachea, incomplete cartilaginous tracheal ring and stenosis of the left main bronchus [5]. Cardiovascular anomalies consist of atrial septal defect, ventricular septal defect, patent ductus arteriosus, tetralogy of Fallot, aortic stenosis, persistent left superior vena cava, common ventricle, and aortic arch anomalies. [6]. Gastrointestinal, genitourinary, vertebral and thyroid anomalies have also been reported. [5]

Half of the affected infants have symptoms at birth and two thirds, by one month of age [6]. Common symptoms are intermittent cyanosis, dyspnoea and apnoeic spells. Inspiratory stridor or expiratory wheeze may be present.

On a CT angiogram, the continuity of anomalous left pulmonary artery from the right pulmonary artery at its origin can be seen as it courses between the trachea and the esophagus to enter the left hilum. This may cause a compression and/or displacement of the trachea and esophagus

The patient also had an ASD, which was detected on echocardiography. Another finding in this case was an anomalous artery arising from the abdominal aorta and supplying the medial basal segment of right lower lobe of the lung, raising the possibility of a sequestered segment. However, the lung parenchyma supplied by the artery appeared normal on the lung windows. An intralobar sequestration consists of an abnormal segment of lung tissue that shares the visceral pleural covering of an otherwise normal pulmonary lobe and that lacks a normal communication to the tracheobronchial tree, although variable amounts of air may be contained within the anomalous tissue. Intralobar sequestrations almost always occur within the lower lobes and occur slightly more often in the left lung. An anomalous systemic artery may supply an area of otherwise normal lung tissue in the absence of congenital heart and lung disease. This condition differs from classic bronchopulmonary sequestration in that the involved lung tissue retains a normal connection to the bronchial tree, although some authors place this entity within the broad framework of pulmonary sequestration. [7]

The aberrant vessel from the abdominal aorta was then embolised which relieved some of the child's symptoms. But the oxygen saturation values still remained low. The patient is awaiting surgery at the time of submitting this report.

Pulmonary artery sling is a rare but a fatal congenital anomaly, which usually calls for immediate surgical intervention in most cases. However, some cases remain asymptomatic and are detected incidentally in asymptomatic adults [5]. Owing to its orientation, the abnormal vessel can be completely hidden behind the proximal part of the right pulmonary artery on a frontal pulmonary angiogram. [8]

Spiral CT angiography is a one-stop shop that has the advantage of avoiding the invasive investigations like bronchoscopy, tracheobronchography and pulmonary artery angiography .A precise diagnosis of both vascular and bronchial anomalies can be obtained in a single data set.

To our knowledge, this is the first reported case of Pulmonary artery sling accompanied by an aberrant vessel of aortic origin supplying the lung parenchyma, with possibly a sequestered medial basal segment of the right lower lobe.

 
   References Top

1.Glaevecke, Doehle.Uber eine selten angeborene Anomalie der Pulmonalarteren. MMW 1897; 44:950-953  Back to cited text no. 1    
2.Capitanio MA, Ramos R, Kirkpatrik JA. Pulmonary sling: roentgen observations. AJR 1971; 112:28-31.  Back to cited text no. 2    
3.Procacci C, Residori E, Bertocco Met al.Left pulmonary artery sling in the adult: case report and review of literature. Cardiovasc Intervent Radiol 1993; 16: 388-391.  Back to cited text no. 3    
4.Arey JB. Malformations of the aorta and aortic arches. In : Arey JB,editor.Cardiovascular pathology in infants and children. WB Saunders Company.1984: 242-4  Back to cited text no. 4    
5.Jue KL, Raghib G, Amplatz K, Adams P Jr, Edwards JE. Anomalous origin of left pulmonary artery from right pulmonary artery. AJR 1965; 95: 598-610  Back to cited text no. 5  [PUBMED]  
6.Sade RM, Rosenthal A, Fellows K, Castenada AR. Pulmonary artery sling. J Thorac Cardiovas Surg 1975; 69:333-346  Back to cited text no. 6    
7.Kyung-Hyun Do, Jin Mo Goo, Jung-Gi Im et al. Systemic Arterial Supply to the Lungs in Adults: Spiral CT Findings. Radiographics.2001;21:387-402.  Back to cited text no. 7    
8.Becker CL, IdrissFS, Holinger LD et al. Pulmonary artery sling: results of surgical repair in infancy. J Thorac Cardiovasc surg 1992; 103: 683-691  Back to cited text no. 8    

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Correspondence Address:
S B Desai
Head of Department of CT, MRI and DSA , Jaslok Hospital and Research Center, Mumbai- 400026
India
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Source of Support: None, Conflict of Interest: None


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    Figures

[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5], [Figure - 6]

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