Hostname: page-component-848d4c4894-ttngx Total loading time: 0 Render date: 2024-06-08T22:35:32.913Z Has data issue: false hasContentIssue false
  • English
  • Français

Duplicated left pulmonary artery: an unknown disease? Three case reports and review of the literature

Published online by Cambridge University Press:  05 March 2015

Valentina Giudici ,
Mazyar Kanani ,
Nagarajan Muthialu ,
Michelle Carr ,
Alistair D. Calder ,
Catherine M. Owens ,
Andrew C. Cook  and
Jan Marek
Valentina Giudici*
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom Paediatric Department Hospital Luigi Sacco, University of Milan, Milan, Italy
Mazyar Kanani
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom
Nagarajan Muthialu
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom
Michelle Carr
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom
Alistair D. Calder
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom
Catherine M. Owens
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom
Andrew C. Cook
Affiliation:
Institute of Cardiovascular Sciences, University College London, London, United Kingdom
Jan Marek
Affiliation:
Great Ormond Street Hospital for Children, London, United Kingdom Institute of Cardiovascular Sciences, University College London, London, United Kingdom
*
Correspondence to: Dr V. Giudici, Paediatric Registrar, Paediatric Department Hospital Luigi Sacco, University of Milan, Via G.B. Grassi 74, 200174 Milan, Italy. Tel: +39 0239042253; Fax: +39 0239042254; E-mail: valentinagiudici@hotmail.it
Get access Rights & Permissions [Opens in a new window]

Abstract

We report three cases of an abnormal finding of duplicated left pulmonary artery: two of these occurring in children with Kabuki syndrome and configuring the setting of a pseudo-pulmonary sling without any clinical or cardiac cross-sectional evidence of tracheal compression. The other case instead represents duplicated left pulmonary artery with pulmonary sling caused by the retro-tracheal course of the lower left pulmonary artery associated with “Christmas Tree” arrangement of the tracheo-bronchial system.

In both patients with pseudo-pulmonary sling and Kabuki syndrome, the abnormal finding was incidental during echocardiographic examination and neither of the patients required surgical repair for the condition. To the best of our knowledge, they represent the third and fourth cases in which such an anomaly of the pulmonary artery branches not forming a sling is seen in association with Kabuki syndrome. Another case represents our second experience and the second case reported in literature with duplicated left pulmonary artery in the setting of a complex tracheal anatomy. In this symptomatic patient, surgical repair of atrial septal defect and relief of the vascular ring were indicated, and the surgical repair was performed successfully at the age of 3 years.

Keywords

Kabuki syndromeaccessory left pulmonary arteryanomalous left pulmonary arterypulmonary artery sling
Type
Original Articles
Information
Cardiology in the Young , Volume 26 , Issue 2 , February 2016 , pp. 340 - 346
Check for updates
Copyright
© Cambridge University Press 2015 

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

1. Erickson, LC, Cocalis, MW, George, L. Partial anomalous left pulmonary artery: new evidence on the development of the pulmonary artery. Pediatr Cardiol 1996; 17: 319–321.Google Scholar
2. Abelardo, E, Hewitt, R, Elliott, MJ, Muthialu, N. Successful surgical repair of complex Christmas tree pattern tracheo bronchial anatomy with stenosis. Eur Arch Otorhinolaryngol 2013; 270: 2161–2163.Google Scholar
3. Digilio, MC, Marino, B, Toscano, A, Giannotti, A, Dallapiccola, B. Congenital heart defects in Kabuki syndrome. Am J Med Genet 2001; 100: 269–274.Google Scholar
4. Jue, KL, Raghib, G, Amplatz, K, Adams, P, Ewards, JE. Anomalous origin of left pulmonary artery from the right pulmonary artery. Am J Roentgenol Radium Ther Nucl Med 1965; 95: 598–610.CrossRefGoogle ScholarPubMed
5. Landing, BH. Five syndromes (malformation complexes) of pulmonary asymmetry, congenital heart disease and multiple spleens. Pediatr Pathol 1984; 2: 125–127.CrossRefGoogle Scholar
6. Nikawa, N, Matsuura, N, Fukushima, Y, et al. Kabuki makeup syndrome: a syndrome of mental retardation, unusual facies, large and protruding ears and postnatal growth deficiency. J Pediatr 1981; 99: 565–569.Google Scholar
7. Kuroki, Y, Suzuki, Y, Chyo, H, et al. A new malformation syndrome of long palpebral fissures, large ears, depressed nasal tip and skeletal anomalies associated with postnatal dwarfism and mental retardation. J Pediatr 1981; 99: 571–573.CrossRefGoogle ScholarPubMed
8. Ohdo, S, Madokoro, H, Sonoda, T, et al. Kabuki makeup syndrome associated with congenital heart disease. J Med Gen 1985; 22: 126–127.Google Scholar
9. Bhat, A, Davenport, J, Cocalis, M. Partial anomalous left pulmonary artery along with aortic coarctation in an infant with Kabuli syndrome. Echocardiography 2012; 29: E145–E147.Google Scholar
10. Collins, RT, Weinberg, PM, Goldmuntz, E, et al. Partial anomalous left pulmonary artery. Circulation 2009; 119: 2405–2407.Google Scholar
11. Bamman, JL, Ward, BH, Woodrum, DE. Aberrant left pulmonary artery, clinical and embryologic factors. Chest 1977; 71: 1.Google Scholar
12. Fountain-Dommer, R, Shirali, G, Wiles, H, Larsen, R. Noninvasive diagnosis of partial anomalous left pulmonary artery. Am J Echocardiogr 2001; 14: 745–746.CrossRefGoogle ScholarPubMed
13. Ge, S, DeGroff, C, Knudson, O, Strain, J, Chan, KC. Noninvasive assessment of pseudo pulmonary artery sling by echocardiography and computerized tomography. Circulation 2001; 103: e115–e116.Google Scholar