Hostname: page-component-7b7567c8c-tnzcf Total loading time: 0 Render date: 2026-11-12T07:50:21.803Z Has data issue: false hasContentIssue false
  • English
  • Français

Late manifestation coarctation of the aorta in a premature infant 4-month post-percutaneous device closure of a patent ductus arteriosus

Part of:Interventional CardiologyAortic Coarctation

Published online by Cambridge University Press:  25 November 2019

Ryan M. SerranoOpen the ORCID record for Ryan M. Serrano[Opens in a new window],
Mark D. Rodefeld and
Ryan Alexy
Ryan M. Serrano*
Affiliation:
Department of Pediatric Cardiology and Department of Pediatric Cardiothoracic Surgery, Riley Hospital for Children, Indianapolis, IN, USA
Mark D. Rodefeld
Affiliation:
Department of Pediatric Cardiology and Department of Pediatric Cardiothoracic Surgery, Riley Hospital for Children, Indianapolis, IN, USA
Ryan Alexy
Affiliation:
Department of Pediatric Cardiology and Department of Pediatric Cardiothoracic Surgery, Riley Hospital for Children, Indianapolis, IN, USA
*
Author for correspondence: R. M. Serrano, Department of Pediatric Cardiology, Riley Hospital for Children, 705 Riley Hospital Drive, RR 127 Indianapolis, IN 46202, USA. Tel: (317) 274 8906; Fax: (317) 274-4022; E-mail: ryserran@iupui.edu
Rights & Permissions [Opens in a new window]

Abstract

Patent ductus arteriosus is the most common cardiovascular abnormality in premature infants. With newly available percutaneous devices, centres are reporting high rates of success and favourable safety profiles with percutaneous closure of haemodynamically significant ductus arteriosi in infants under 1000 g. We report the case of a 5-week-old, previous 25-week gestation, 1200-g infant who underwent successful percutaneous closure of a ductus arteriosus with a Medtronic Microvascular Plug but who developed late-term coarctation from the device. This case should prompt practitioners to consider the need and timing of follow-up echocardiograms in this population and sheds light on a newly reported long-term complication of device closure in premature infants.

Keywords

Patent ductus arteriosuscoarctation of the aortaprematurity

Information

Type
Brief Report
Information
Cardiology in the Young , Volume 29 , Issue 12 , December 2019, pp. 1556 - 1558
Copyright
© Cambridge University Press 2019 

A patent ductus arteriosus is present in nearly two-thirds of extremely low-birth-weight infants.Reference Chiruvolu, Punjwani and Ramaciotti1 Classically, practitioners have debated whether or not we should be treating these at all,Reference Noori, Friedlich and Seri2 but when treatment is deemed necessary, the debate has shifted to the pros and cons of surgical ligation versus percutaneous device closure. With newer devices and novel approaches, percutaneous closure in infants less than 1000 g is being completed more frequently. The results have been promising, with minimal morbidity and mortality and potentially less short-term post-operative haemodynamic instability.Reference Ogando, Asensio and Sanchez de la blanca3 We report the case of an uncomplicated percutaneous closure of a patent ductus in a 1200-g infant who developed a coarctation of the aorta 4-month post-procedure.

Case report

Our patient was born at 25-week and 3-day gestation with a birth weight of 650 g. At 1 month, an echocardiogram showed a large ductus arteriosus and severe pulmonary overcirculation. In light of these findings and the fact that was unable to wean from the high-frequency oscillator, the decision was made to proceed with percutaneous closure.

At the time of the procedure, the patient weighed 1200 g. Venous access was obtained with a four French sheath using ultrasound guidance. The duct measured 9.4 mm long and 3.1 mm at the narrowest diameter. A size 5 Medtronic microvascular plug was positioned so that the distal “uncovered” portion of the device was within the lumen of the aorta. Transthoracic echocardiography performed immediately before and after device release showed no residual shunting and no obstruction to flow in the left pulmonary artery or the descending aorta. Follow-up echocardiograms were performed at 1 day, 1 week, and 1 month after closure. They all demonstrated the device in appropriate position without obstruction to flow in the pulmonary artery or aorta (Fig 1a and b). The aortic isthmus measured 3.8 mm (z score 1.4).

Figure 1.

Aortic arch with colour immediately post-patent ductus arteriosus device closure showing a wide open arch (a) and a normal Doppler pattern 1-month post-procedure (b). Echo 4-month post-procedure with colour shows aliasing at the site of coarctation (c) and 3-m/s velocity and diastolic antegrade flow continuation (d).

Four-month post-device closure, an echocardiogram was repeated to assess a new murmur. While the device appeared in the appropriate position, there was a moderate narrowing of the descending aorta adjacent to the device with a peak gradient of 35 mmHg (Fig 1c and d). Pulses were described as 1+ in all extremities, and brachiofemoral delay was not documented. He was normotensive; however, the only blood pressures recorded were taken in the left lower extremity. This was thought to represent true native coarctation rather than obstruction from the device given the serial appearance by echocardiography and the mild distal displacement of the left subclavian artery. A CT scan confirmed a moderate-to-severe coarctation in the juxtaductal region with mild displacement of the left subclavian artery; however, contrast did not fill the uncovered portion of the device, which extended into the aorta (Fig 2a and b). The aortic isthmus measured 2 mm (z score −2.99).

Figure 2.

3-D reconstruction and CT scan showing moderate-to-severe coarctation (a and b). Path specimen showing the aortic end of the Medtronic microvascular plug has endotheliased and fibrosed to the posterior aspect of the aortic wall and has formed a double-barrel appearance within the aortic lumen (c and d).

The patient underwent an extended end-to-end repair of the coarctation at 6 months of age. Prior to repair, a proximal right radial arterial pressure was 92/46 compared to a distal lower extremity femoral arterial line pressure of 68/45. The coarctation segment, including half of the device, was resected. Post-operatively, there was no residual gradient from the upper to lower extremities. The aortic wall specimen appeared thick and fibrotic, consistent with a native coarctation. The “uncovered” portion of the device was completely endothelialised. The distal pin of the device appeared to be fibrosed and adherent to the posterior wall of the aorta, creating a double-barrel lumen in the region of the coarctation (Fig 2c and d). Histopathology showed fibrosis consistent with coarctation; however, the pathologist could not determine the likelihood of a reactive fibrosis. Post-operative echo showed a widely patent aortic arch.

Discussion

Over the last several years, centres have reported success using novel devices and techniques for percutaneous closure of a patent ductus arteriosus in infants under 1 kg. The major risks reported have been limited to events that occur during or shortly post-procedure (<24 hours) and include device embolisation, vessel damage, atrial perforation, left pulmonary artery stenosis, and iatrogenic aortic coarctation.Reference Wang-Giuffre and Breinholt4–Reference Zahn, Peck and Phillips7 Despite an angiogram of the aorta during the catheterisation procedure and all subsequent echocardiograms demonstrating a patent aorta up to 1 month following the procedure, our patient developed a significant late coarctation.

This may have been the result of a true native coarctation secondary to late constriction of ductal tissue extending into the aorta, which is supported by the mildly displaced left subclavian artery, the posterior shelf seen on the CT, and gross examination of the aortic specimen. Anecdotally, our surgeons report having seen development of late native coarctation in premature infants after surgical patent ductus arteriosus ligation, so it is reasonable to assume that it can develop after percutaneous device closure in the same population. An alternative explanation is that this was the result of a reactive fibrosis of the aorta triggered by the device. This explanation seems unlikely to completely explain our findings given the frequency with which patent ductus arteriosus devices extend into the aorta and the infrequency with which we see coarctations develop after device closure.

The most surprising finding was the complete endothelialisation of the uncovered distal portion of the device. Previously, it was assumed that blood would flow unobstructed through the uncovered wire at the distal portion of the device and down the descending aorta. In this case, however, the “wire barrel” was completely covered with endothelium, preventing flow through the device. It is unclear if endothelialisation is the natural progression of all Medtronic microvascular plugs or if this was a reactive process triggered by an abnormal aortic wall.

Until now, our experience with over 25 premature infants having undergone percutaneous ductal closure has also been limited to in-procedure morbidity, with only one patient experiencing device embolisation that was successfully retrieved without further complication. Our standard post-procedure protocol has included an echocardiogram at 1-day, 1-week, 1-month, and 1-year post-closure, but this case highlights the importance of closer follow-up for these patients. Our institutional post-procedural protocol now includes an additional echocardiogram 4–6 months after device closure.

Conclusion

Practitioners should be aware of the potential for late presentation of coarctation of the aorta in premature infants who have undergone ductal device closure with the Medtronic microvasular plug and should consider evaluating these patients 4- to 6-month post-procedure.

Acknowledgements

The authors would like to acknowledge Dr John J. Parent for his assistance in editing the manuscript.

Financial Support

This research received no specific grant from any funding agency, commercial, or not-for-profit sectors.

Conflicts of Interest

None.

Ethical Standards

The authors have no relationships or interests that could have direct or potential influence or impart bias on the work to disclose. No research was conducted involving patients and/or animals. The authors assert that all procedures contributing to this work comply with the ethical standards of the relevant national guidelines on human experimentation and with the Helsinki Declaration of 1975, as revised in 2008, and has been approved by the institutional committee.

Contributor’s Statement

Dr R.M.S. drafted the initial manuscript, revised the manuscript, and approved the final manuscript as submitted. Dr M.D.R. reviewed and revised the manuscript and approved the final manuscript as submitted. Dr R.A. reviewed and revised the manuscript and approved the final manuscript as submitted.

References

Chiruvolu, A, Punjwani, P, Ramaciotti, C. Clinical and echocardiographic diagnosis of patent ductus arteriosus in premature neonates. Early Hum Dev 2009; 85: 147–149.CrossRefGoogle ScholarPubMed
Noori, S, Friedlich, P, Seri, I. Patent ductus arteriosus in the preterm infant: to treat or not to treat? J Perinatol 2010; 30: S31–S37.CrossRefGoogle ScholarPubMed
Ogando, AR, Asensio, IP, Sanchez de la blanca, AR, et al. Surgical ligation versus percutaneous closure of patent ductus arteriosus in very low-weight preterm infants: which are the real benefits of the percutaneous approach? Pediatr Cardiol 2018; 39: 398–410.CrossRefGoogle Scholar
Wang-Giuffre, EW, Breinholt, J. Novel use of the Medtronic micro vascular plug for PDA closure in preterm infants. Catheter Cardiovasc Interv 2017; 89: 1059–1065.CrossRefGoogle ScholarPubMed
Sathanandam, S, Justino, H, Warrer, BR 3rd, et al. Initial clinical experience with the Medtronic Micro Vascular Plug in transcatheter occlusion of PDAs in extremely premature infants. Catheter Cardiovasc Interv 2017; 89: 1051–1058.CrossRefGoogle ScholarPubMed
Pamukcu, O, Tuncay, A, Narin, N, et al. Patent ductus arteriosus closure in preterms less than 2 kg: surgery versus transcatheter. Int J Cardiol 2018; 250: 110–115.CrossRefGoogle ScholarPubMed
Zahn, EM, Peck, D, Phillips, A, et al. Transcatheter closure of patent ductus arteriosus in extremely premature newborns early results and midterm-follow up. J Am Coll Cardiol Intv 2016; 9: 2429–2437.CrossRefGoogle ScholarPubMed
Figure 0

Figure 1.Aortic arch with colour immediately post-patent ductus arteriosus device closure showing a wide open arch (a) and a normal Doppler pattern 1-month post-procedure (b). Echo 4-month post-procedure with colour shows aliasing at the site of coarctation (c) and 3-m/s velocity and diastolic antegrade flow continuation (d).

Figure 1

Figure 2.3-D reconstruction and CT scan showing moderate-to-severe coarctation (a and b). Path specimen showing the aortic end of the Medtronic microvascular plug has endotheliased and fibrosed to the posterior aspect of the aortic wall and has formed a double-barrel appearance within the aortic lumen (c and d).