Pulmonary arteriovenous malformation as a cause of exercise intolerance in children: a case report

  • Pierluigi Morreale
  • Veronica Notarbartolo Università degli Studi di Palermo
  • Giancarlo Allegro
  • Mario Giuseppe Vallone
  • Francesca Finazzo
  • Vincenzo Antona
  • Maria Fiorella Sanfilippo
  • Calogero Comparato
Keywords: pulmonary arteriovenous malformation; exercise intolerance; oxygen desaturation; case-report

Abstract

Pulmonary arteriovenous malformations (PAVMs) in children are rare lesions characterized by abnormal low resistance vascular structures connecting a pulmonary artery to a pulmonary vein, resulting in an intrapulmonary right-to-left shunt. The insidious onset and variable signs and symptoms make diagnosis difficult, especially in children. PAVMs can be single or multiple, congenital or acquired, and up to 47-80% of cases are associated with hereditary hemorrhagic telangiectasia (HHT).

References

1. Saboo SS, Chamarthy M, Bhalla S, Park H, Sutphin P, Kay F, et al. Pulmonary arteriovenous malformations: diagnosis. Cardiovasc Diagn Ther. 2018;8:325-37. DOI: https://doi.org/10.21037/cdt.2018.06.01.
2. Mowers KL, Sekarski L, White AJ, Grady RM. Pulmonary arteriovenous malformations in children with hereditary hemorrhagic telangiectasia: a longitudinal study. Pulm Circ. 2018;8:2045894018786696. DOI: https://doi.org/10.1177/2045894018786696.
3. Feinstein JA, Moore P, Rosenthal DN, Puchalski M, Brook MM. Comparison of contrast echocardiography versus cardiac catheterization for detection of pulmonary arteriovenous malformations. Am J Cardiol. 2002;89:281-5. DOI: https://doi.org/10.1016/s0002-9149(01)02228-7.
4. Chang RK, Alejos JC, Atkinson D, Jensen R, Drant S, Galindo A, et al. Bubble contrast echocardiography in detecting pulmonary arteriovenous shunting in children with univentricular heart after cavopulmonary anastomosis. J Am Coll Cardiol. 1999;33:2052-8. DOI: https://doi.org/10.1016/s0735-1097(99)00096-0.
5. Shovlin CL. Pulmonary arteriovenous malformations. Am J Respir Crit Care Med. 2014;190:1217-28. DOI: https://doi.org/10.1164/rccm.201407-1254CI.
6. Rotondo E, Sgrazzutti L, Attanasi M, Petrosino MI, Chiarelli F, Di Pillo S. When polycythemia and exertional dyspnea are not asthma. Pneumologia Pediatrica. 2019;19:21-5. Www.simri.it. Accessed on 05, 2nd 2023.7)
7. Khurshid I, Downie GH. Pulmonary arteriovenous malformation. Postgrad Med J. 2002;78:191-7. DOI: https://doi.org/10.1136/pmj.78.918.191.
8. Wong HH, Chan RP, Klatt R, Faughnan ME. Idiopathic pulmonary arteriovenous malformations: clinical and imaging characteristics. Eur Respir J. 2011;38:368-75. DOI: https://doi.org/10.1183/09031936.00075110.
9. Sladden D, Casha A, Azzopardi C, Manchè A. A large pulmonary arteriovenous malformation causing cerebrovascular accidents. BMJ Case Rep. 2015;2015:bcr-2014207786. DOI: https://doi.org/10.1136/bcr-2014-207786.
10. Meek ME, Meek JC, Beheshti MV. Management of pulmonary arteriovenous malformations. Semin Intervent Radiol. 2011;28:24-31. DOI: https://doi.org/10.1055/s-0031-1273937.
Published
2023-06-27
How to Cite
1.
Morreale P, Notarbartolo V, Allegro G, Vallone M, Finazzo F, Antona V, Sanfilippo M, Comparato C. Pulmonary arteriovenous malformation as a cause of exercise intolerance in children: a case report. PI [Internet]. 27Jun.2023 [cited 27Apr.2024];63(3):208-2. Available from: https://paediatricaindonesiana.org/index.php/paediatrica-indonesiana/article/view/2637
Section
Case Report
Received 2021-03-31
Accepted 2023-06-27
Published 2023-06-27