Hostname: page-component-78c5997874-4rdpn Total loading time: 0 Render date: 2024-11-10T06:45:50.179Z Has data issue: false hasContentIssue false

Repair of isolated atrial septal defect in infants less than 12 months improves symptoms of chronic lung disease or shunt-related pulmonary hypertension

Published online by Cambridge University Press:  16 March 2020

Dafni Charisopoulou*
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK Department of Pediatrics, Division of Pediatric Cardiology, Amalia Children's Hospital, Radboud University Medical Center, Nijmegen, Netherlands Academic Centre for Congenital Heart Disease, Netherlands
Roberta Margarita Bini
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK
Gillian Riley
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK
Kalai Janagarajan
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK
Shahin Moledina
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK Institute of Cardiovascular Science, UCL, London, UK
Jan Marek
Affiliation:
Cardiorespiratory Unit, Great Ormond Street Hospital for Children, London, UK Institute of Cardiovascular Science, UCL, London, UK
*
Author for correspondence: Dr D. Charisopoulou, Department of Pediatrics, Division of Pediatric Cardiology, Radboud University Medical Center, 6500 HC Nijmegen, Netherlands. Tel: +0044651912675. E-mail: dafnithess@yahoo.co.uk

Abstract

Introduction:

Infants with isolated atrial septal defects are usually minimally symptomatic, and repair is typically performed after infancy. Early repair may be considered if there is high pulmonary blood flow and reduced respiratory reserve or early signs of pulmonary hypertension. Our aim was to review the characteristics and outcomes of a cohort of patients who underwent infant repair at our institute.

Methods:

The study included 56 infants (28 female, 19 trisomy 21) with isolated atrial septal defect (age: 8 months (1.5–12), weight: 6 kg (2.8–7.5), echo Qp/Qs: 1.9 ± 0.1) who underwent surgical closure (20 fenestrated). Three groups were identified: 1) chronic lung disease and pulmonary hypertension (group A: n = 28%); 2) acutely unwell infants with pulmonary hypertension but no chronic lung disease (group B: n = 20, 36%); and 3) infants with refractory congestive heart failure without either pulmonary hypertension or chronic lung disease (group C: n = 9, 16%).

Results:

Post-operatively, pulmonary hypertension infants (47/56) showed improvement in tricuspid annular plane systolic excursion z-score (p < 0.001) and right ventricular systolic/diastolic duration ratio (p < 0.05). All ventilator (14.3%) or oxygen-dependent (31.6%) infants could be weaned within 2 weeks after repair. One year later, weight z-score increased in all patients and by +1 in group A, +1.3 in group B and +2 in group C. Over a median follow-up of 1.4 years, three patients died, four patients continued to have pulmonary hypertension evidence and two remained on targeted pulmonary hypertension therapy.

Conclusion:

Atrial septal defect repair within the first year may improve the clinical status and growth in infants with early signs of pulmonary hypertension or those requiring respiratory support and facilitate respiratory management.

Type
Original Article
Copyright
© The Author(s), 2020. Published by Cambridge University Press

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

Choi, EK, Jung, YH, Kim, HS, et al.The impact of atrial left-to-right shunt on pulmonary hypertension in preterm infants with moderate or severe bronchopulmonary dysplasia. Pediatr Neonatol 2015 October; 56: 317323.CrossRefGoogle ScholarPubMed
Hascoet, JM, Hamon, I, Didier, F, Maurer, P, Vert, P. Patent foramen ovale with left to right shunt in bronchopulmonary dysplasia: coincidental or associated complication? Acta Paediatr 1994; 83: 258261.CrossRefGoogle ScholarPubMed
Lim, DS, Matherne, GP (2007) Percutaneous device closure of atrial septal defect in a premature infant with rapid improvement in pulmonary status. Pediatrics 119: 3984001.CrossRefGoogle Scholar
Deddy, NR, Chilakala, SR, Rana, D. Anatomical closure of left-to-right shunts in premature infants with bronchopulmonary dysplasia and pulmonary hypertension: a cautionary tale. AJP Reports 2015 October; 5: e097e098.Google Scholar
Chen, Q, Cao, H, Zhang, GC, Chen, LW, Xu, F, Zhang, JX. Short term and mid term follow-up of transthoracic device closure of atrial septal defect in infants. Ann Thorac Surg 2017 October; 104: 14031409.CrossRefGoogle Scholar
Thomas, VC, Vincent, R, Raviele, A, Diehl, H, Qian, H, Kim, D. Transcatheter closure of secundum atrial septal defect in infants less than 12 months of age improves symptoms of chronic lung disease. Congenit Heart Dis 2012 May-June; 7: 204211.CrossRefGoogle ScholarPubMed
Wood, AM, Holzer, RJ, Texter, KM, et al.Transcatheter elimination of left-to-right shunts in infants with bronchopulmonary dysplasia is feasible and safe. Congenit Heart Dis 2011 July-August; 6: 330337.CrossRefGoogle ScholarPubMed
Goetschmann, S, Dibernardo, S, Steinmann, H, Pavlovic, M, Sekarski, N, Pfammatter, JP. Frequency of severe pulmonary hypertension complicating “isolated” atrial septal defect in infancy. Am J Cardiol 2008 August 1; 102: 340342.CrossRefGoogle ScholarPubMed
Diab, KA, Cao, QL, Bacha, EA, Hijazi, ZM. Device closure of atrial septal defects with the Amplatzer septal occluder: safety and outcome in infants. J Thoracic Cardiovasc Surg 2007; 134: 960966.CrossRefGoogle ScholarPubMed
Lammers, A, Hager, A, Eicken, A, Lange, R, Hauser, M, Hess, J. Need for closure of secundum atrial septal defect in infancy. J Thoracic Cardiovasc Surg 2005; 129: 13531357.CrossRefGoogle ScholarPubMed
Andrews, R, Tulloh, R, Magee, A, Anderson, D. Atrial septal defect with failure to thrive in infancy: hidden pulmonary vascular disease? Pediatr Cardiol 2002 September- October; 23: 528530.CrossRefGoogle ScholarPubMed
Bull, C, Deanfield, J, de Leval, M, Stark, J, Taylor, JF, Macartney, FJ. Correction of isolated secundum atrial septal defect in infancy. Arch Dis Child 1981 October; 56: 784786.CrossRefGoogle ScholarPubMed
Silvestry, FE, Cohen, MS, Armsby, LB, et al.Guidelines for the echocardiographic assessment of atrial septal defect and patent foramen ovale: from the American Society of Echocardiography and Society for Cardiac Angiography and Interventions. J Am Soc Echocardiogr 2015; 28: 910958.CrossRefGoogle ScholarPubMed
Rudski, LG, Lai, WW, Afilalo, J, et al.Guidelines for the echocardiographic assessment of the right heart in adults: a report from the American Society of Echocardiographyendorsed by the European Association of Echocardiography, a registered branch of the European Society of Cardiology, and the Canadian Society of Echocardiography. J Am Soc Echocardiogr 2010 July; 23: 685713; quiz 786–788.CrossRefGoogle Scholar
McCrary, A, Malowitz, JR, Hornick, CP, et al.Differences in eccentricity index and systolic-diastolic ratio in extremely low-birth-weight infants with bronchopulmonary dysplasia at risk of pulmonary hypertension. Am J Perinatol 2016 January; 33: 5762.CrossRefGoogle ScholarPubMed
McQuillan, BM, Picard, MH, Leavitt, M, Weyman, AE. Clinical correlates and reference intervals for pulmonary artery systolic pressure among echocardiographically normal subjects. Circulation 2001; 104: 27972802.CrossRefGoogle ScholarPubMed
Farquhar, M, Fitzgerald, DA. Pulmonary hypertension in chronic neonatal lung disease. Paediatr Respir Rev 2010; 11: 149153.CrossRefGoogle ScholarPubMed
Ambalavanan, N, Mourani, P. Pulmonary hypertension in bronchopulmonary dysplasia. Birth Defects Res A Clin Mol Teratol 2014; 100: 240246.CrossRefGoogle ScholarPubMed
Bossone, E, D’Andrea, A, D’Alto, M, et al.Echocardiography in pulmonary arterial hypertension: from diagnosis to prognosis. J Am Soc Echocardiogr 2013 January; 26: 114.CrossRefGoogle ScholarPubMed
Faltering Growth – recognition and management. National Institute for Health and Care Excellence: Clinical Guidelines. London: National Institute for Health and Care Excellence (UK). ISBN 9781473126930. PMID 28991420.Google Scholar
Kinsella, JP, Greeough, A, Abman, SH. Bronchopulmonary dysplasia. Lancet 2006 April 29; 367: 14211431.CrossRefGoogle ScholarPubMed
Mainwaring, RD, Mirali-Akbar, H, Lamberti, JJ, Moore, JW. Secundum-type atrial septal defects with failure to thrive in the first year of life. J Card Surg 1996; 11: 116120.CrossRefGoogle ScholarPubMed
Wyss, Y, Quandt, D, Weber, R, et al.Interventional closure of secundum type atrial septal defects in infants less than 10 kilograms: indications and procedural outcome. J Interv Cardiol 2016 December; 29: 646653.CrossRefGoogle ScholarPubMed
Del Cerro, ML, Sabaté Rotés, A, Cartón, A, et al.Pulmonary hypertension in bronchopulmonary dysplasia: clinical findings, cardiovascular anomalies and outcomes. Pediatr Pulmonol 2014; 49: 4959.CrossRefGoogle ScholarPubMed
Cantinotti, M, Assanta, N, Murzi, B, Lopez, L. Controversies in the definition and management of insignificant left-to-right shunts. Heart 2014; 100: 200205.CrossRefGoogle ScholarPubMed
Lee, C, Lim, G, Kim, WS, Han, HS. Clinical characteristics and outcome of incidental atrial septal openings in very low birth weight infants. Neonatology 2014; 105: 8590.CrossRefGoogle ScholarPubMed
Evans, N, Iyer, P. Incompetence of the foramen ovale in preterm infants supported by mechanical ventilation. J Pediatr 1994; 125: 786792.CrossRefGoogle ScholarPubMed
Motz, R, Grassl, G, Trawoger, R. Dependence on a respiratory ventilator due to an atrial septal defect. Cardiol Young 2000; 10: 150152.CrossRefGoogle Scholar
Chess, PR, D’Angio, CT, Pryhuber, GS, Maniscalco, WM. Pathogenesis of bronchopulmonary dysplasia. Semin Perinatol 2006; 30: 171178.CrossRefGoogle ScholarPubMed
Kumar, VH, Hutchison, AA, Lakshminrusimha, S, Morin, FC 3rd, Wynn, RJ, Ryan, RM. Characteristics of pulmonary hypertension in preterm neonates J Perinatol 2007; 27: 214219.CrossRefGoogle ScholarPubMed
Rhee, EK, Evangelista, JK, Nigrin, DJ, et al.Impact of anatomic closure on somatic growth among small, asymptomatic children with secundum atrial septal defect. Am J Cardiol 2000; 85: 14721475.CrossRefGoogle ScholarPubMed
Bishnoi, RN, Everett, AD, Ringel, RE, et al.Device closure of secundum atrial septal defects in infants weighing less than 8 kg. Pediatr Cardiol 2014 October; 35: 11241131.CrossRefGoogle ScholarPubMed
Malowitz, JR, Forsha, DE, Smith, PB, Cotten, CM, Barker, PC, Tatum, GH. Right ventricular echocardiographic indices predict poor outcomes in infants with persistent pulmonary hypertension of the newborn. Eur Heart J Cardiovasc Imaging. 2015 November; 16: 12241231.CrossRefGoogle ScholarPubMed
Lewandowski, AJ, Bradlow, WM, Augustine, D, et al.Right ventricular systolic dysfuction in young adults born preterm. Circulation 2013; 128: 714720.CrossRefGoogle Scholar