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Resolution of severe pulmonary artery obstruction secondary to ductus arteriosus aneurysm: a case report with long-term follow-up and balloon angioplasty

Published online by Cambridge University Press:  11 August 2025

Takafumi SeoOpen the ORCID record for Takafumi Seo[Opens in a new window],
Takeshi Sasaki and
Hisashi SugiyamaOpen the ORCID record for Hisashi Sugiyama[Opens in a new window]
Takafumi Seo*
Affiliation:
Department of Pediatric Cardiology and Electrophysiology, Osaka City General Hospital, Osaka, Japan
Takeshi Sasaki
Affiliation:
Department of Pediatric Cardiology and Electrophysiology, Osaka City General Hospital, Osaka, Japan
Hisashi Sugiyama
Affiliation:
Department of Pediatric Cardiology and Electrophysiology, Osaka City General Hospital, Osaka, Japan
*
Corresponding author: Takafumi Seo; Email: t.seo16med@gmail.com
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Abstract

Ductus arteriosus aneurysm typically resolves spontaneously; surgical resection is required when it compresses surrounding structures and causes symptoms. In our neonatal case, the ductus arteriosus gradually regressed, but the left pulmonary artery stenosis persisted; therefore, balloon dilation was performed to address the ongoing imbalance in bilateral pulmonary perfusion. This case underscores the potential for non-surgical management of ductus arteriosus aneurysms.

Keywords

Ductus arteriosus aneurysmPulmonary artery stenosisPercutaneous balloon dilation

Information

Type
Case Report
Information
Cardiology in the Young , Volume 35 , Issue 9 , September 2025, pp. 1947 - 1949
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Copyright
© The Author(s), 2025. Published by Cambridge University Press

Introduction

Ductus arteriosus aneurysm is characterised by cystic or tubular dilation of the ductus arteriosus, with an incidence ranging from 0.8% to 8.8%. Reference Falcone, Perloff and Roberts1–Reference Jan, Hwang, Fu, Chai and Chi2 While ductus arteriosus aneurysms typically regress and resolve spontaneously, they may occasionally lead to serious complications such as thromboembolism, infection, rupture, and compression of adjacent structures. Reference Dyamenahalli, Smallhorn and Geva3 Furthermore, ductus arteriosus aneurysm associated with severe pulmonary artery stenosis is rare. Herein, we describe various imaging modalities for neonates with a ductus arteriosus aneurysm and severe pulmonary artery stenosis. We also report the postnatal clinical course of an infant, including successful catheter intervention for pulmonary artery stenosis caused by ductus arteriosus aneurysm.

Case report

A full-term 17-day-old girl was referred to the Paediatric Cardiology Department with a heart murmur. The infant showed no symptoms of respiratory distress. Echocardiography revealed a well-circumscribed echogenic mass (11 mm × 10 mm) extending into the main pulmonary artery (Fig 1a). The mass occupied the distal main pulmonary artery, causing pulmonary artery stenosis in both the main and bilateral branches (peak velocity: 3.9 m/s on the right and 3.5 m/s on the left). Owing to pulmonary artery stenosis resulting from mass compression, the estimated right ventricular pressure was approximately equal to the left ventricular pressure. Blood flow in the left pulmonary artery was significantly lower than that in the right pulmonary artery. Enhanced CT and cardiac MRI revealed a mass lesion. Enhanced CT revealed a 24- × 11- × 18-mm mass lesion without enhancement from the main pulmonary artery trunk to the pulmonary artery bifurcation (Fig 1b). MRI revealed a high-intensity mass on T1 and T2 images (Fig 1c). Based on these findings, ductus arteriosus aneurysm and mediastinal tumours were suspected. At 2 weeks of follow-up, the interior of the mass solidified and started to shrink (Fig 1d). Therefore, the child was diagnosed with ductus arteriosus aneurysm. The reduction in the mass size led to an improvement in the main pulmonary stenosis, allowing for close observation without intervention. Echocardiography at 6 months of age showed that the left pulmonary artery stenosis persisted, whereas the main and right pulmonary artery stenoses completely resolved (Fig 2a). Cardiac MRI performed at 8 months of age revealed a right-to-left pulmonary artery blood flow ratio of five, confirming a significant imbalance in bilateral pulmonary blood flow. Contrast-enhanced CT revealed that the ductus arteriosus aneurysm had sufficiently shrunk, resulting in decompression of the left pulmonary artery (Fig 2b). The cause of the left pulmonary artery stenosis was speculated to be contraction of residual ductal tissue rather than compression by the ductus arteriosus aneurysm. Percutaneous balloon angioplasty for the left pulmonary artery stenosis was performed at 10 months of age. At pre-dilatation, the right ventricular pressure was restored to the normal range, but severe left pulmonary artery stenosis was still evident on angiography (Fig 2c). A 6-mm Sterling™ balloon catheter (Boston Scientific, Natick, MA, USA) dilated the lesion from 3.2 to 5.0 mm, markedly improving the distal left pulmonary artery pressure waveform. At one and a half years of age, echocardiography showed only mild left pulmonary artery stenosis (peak velocity: 1.7 m/s), and the ductus arteriosus aneurysm disappeared completely. At the latest follow-up, the patient remained asymptomatic with normal growth and no evidence of restenosis.

Figure 1.

Different imaging studies performed on the mass lesion at the initial visit and at 1 month of age. ( a ) Parasternal short-axis view of transthoracic echocardiography at 17 days. Image showing a round mass lesion measuring 11 * 10 mm (arrow) at the main pulmonary artery that almost completely obstructs the origin of the left pulmonary artery. ( b ) Cardiac computed tomography with contrast. Axial view ( b1 ) and 3-dimensional reconstructed imaging ( b2 ) showing a 24 * 11 * 18 mm mass (arrow) with no contrast effect. ( c ) Cardiac magnetic resonance imaging. The mass showed high-intensity signals on T1 and T2 images. The differential diagnoses were ductus arteriosus aneurysm (DAA) and mediastinal tumour. ( d ) Parasternal short-axis view of transthoracic echocardiography at 1 month. The mass appeared smaller than that in the neonatal period with a thrombosed lumen. MPA = main pulmonary artery; RPA = right pulmonary artery; LPA = left pulmonary artery.

Figure 2.

Imaging after follow-up for ductus arteriosus aneurysm (DAA) and catheterisation of residual pulmonary artery stenosis. ( a ) Parasternal short-axis view of transthoracic echocardiography at 6 months. The mass almost disappeared, and the main and right pulmonary artery stenoses improved. Mild left pulmonary artery stenosis persisted. ( b ) Cardiac computed tomography image with contrast on the axial view showing that the DAA did not compress the LPA. The LPA stenosis was presumed to result from traction by the patent ductus arteriosus tissues. ( c ) Lateral view of catheter pulmonary artery angiogram and pressure wave forms before dilation ( c1 ) and catheter LPA angiogram after dilation ( c2 ) at 10 months. The LPA was stenosed at the bifurcation (arrow). Balloon angioplasty improved the stenosis and significantly improved the pressure waveform. MPA = main pulmonary artery; RPA = right pulmonary artery; LPA = left pulmonary artery; DsAo = descending aorta.

Discussion

Although the exact incidence rate of ductus arteriosus aneurysm is unclear, it is no longer considered a rare anatomy. In most cases, ductus arteriosus aneurysm resolves spontaneously without symptoms. The complications of ductus arteriosus aneurysm, such as sudden rupture, thromboembolism, infection, bronchial erosion, and surrounding pressure symptoms, are rare. To date, only a few neonatal cases of ductus arteriosus aneurysm with pulmonary artery stenosis have been reported. Reference Dyamenahalli, Smallhorn and Geva3–Reference Takajo and Kobayashi7 Most cases required surgical resection, except for one reported case of isolated left pulmonary artery stenosis that improved spontaneously after 4 months of follow-up. Reference Takajo and Kobayashi7 Determining the optimal management strategy—surgical intervention, catheter intervention, or close observation—remains challenging. A previous report indicated surgery for a ductus arteriosus aneurysm with significant symptoms due to compression of surrounding organs and recommended 4–6 weeks of follow-up to check the spontaneous shrinking of the ductus arteriosus aneurysm. In the present case, echocardiography showed a relatively clear outline of the ductus arteriosus aneurysm, which was associated with a low risk of embolism. The patient was hospitalised and closely monitored for thrombotic symptoms. No oral antiplatelet medications were administered. After 6 months of follow-up, the main pulmonary artery stenosis had completely resolved, but the left pulmonary artery stenosis persisted. As the ductus arteriosus aneurysm had regressed, left pulmonary artery stenosis was attributed to residual patent ductus arteriosus tissue rather than direct compression from the ductus arteriosus aneurysm, prompting balloon dilation. This is the first case report of spontaneous resolution of pulmonary artery stenosis to the extent of nearly complete occlusion of the main pulmonary artery and improved residual left pulmonary artery stenosis with combined balloon dilation. Through careful follow-up, some patients with ductus arteriosus aneurysm can achieve improvement without surgical intervention. While our case demonstrates the potential for conservative management, we do not advocate for routine non-surgical management of all patients with ductus arteriosus aneurysms and severe pulmonary artery stenosis. Each case must be carefully evaluated to determine the most appropriate treatment approach.

Data availability statement

Data sharing does not apply to this article, as no datasets were generated or analysed during the current study.

Acknowledgements

None.

Financial support

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

Competing interests

The authors declare none.

Ethical standard

The authors assert that all procedures contributing to this work comply with the ethical standards of the relevant national guidelines on human experimentation and has been approved by the Institutional Review Board of Osaka City General Hospital.

Patient consent statement

Informed consent was obtained in the form of opt-out on the website of Osaka City General Hospital.

References

Falcone, MW, Perloff, JK, Roberts, WC. Aneurysm of the nonpatent ductus arteriosus. Am J Cardiol 1972; 29: 422–426.CrossRefGoogle ScholarPubMed
Jan, SL, Hwang, B, Fu, YC, Chai, JW, Chi, CS. Isolated neonatal ductus arteriosus aneurysm. J Am Coll Cardiol 2002; 39: 342–347.CrossRefGoogle ScholarPubMed
Dyamenahalli, U, Smallhorn, JF, Geva, T, et al. Isolated ductus arteriosus aneurysm in the fetus and infant: a multi-institutional experience. J Am Coll Cardiol 2000; 36: 262–269.CrossRefGoogle ScholarPubMed
Jeong, L-E, Na, JY, Huh, J, et al. Echogenic mass lesion within the main pulmonary artery in a neonate. Neonatal Med 2020; 27: 89–93.CrossRefGoogle Scholar
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Figure 0

Figure 1.Different imaging studies performed on the mass lesion at the initial visit and at 1 month of age. (a) Parasternal short-axis view of transthoracic echocardiography at 17 days. Image showing a round mass lesion measuring 11 * 10 mm (arrow) at the main pulmonary artery that almost completely obstructs the origin of the left pulmonary artery. (b) Cardiac computed tomography with contrast. Axial view (b1) and 3-dimensional reconstructed imaging (b2) showing a 24 * 11 * 18 mm mass (arrow) with no contrast effect. (c) Cardiac magnetic resonance imaging. The mass showed high-intensity signals on T1 and T2 images. The differential diagnoses were ductus arteriosus aneurysm (DAA) and mediastinal tumour. (d) Parasternal short-axis view of transthoracic echocardiography at 1 month. The mass appeared smaller than that in the neonatal period with a thrombosed lumen. MPA = main pulmonary artery; RPA = right pulmonary artery; LPA = left pulmonary artery.

Figure 1

Figure 2.Imaging after follow-up for ductus arteriosus aneurysm (DAA) and catheterisation of residual pulmonary artery stenosis. (a) Parasternal short-axis view of transthoracic echocardiography at 6 months. The mass almost disappeared, and the main and right pulmonary artery stenoses improved. Mild left pulmonary artery stenosis persisted. (b) Cardiac computed tomography image with contrast on the axial view showing that the DAA did not compress the LPA. The LPA stenosis was presumed to result from traction by the patent ductus arteriosus tissues. (c) Lateral view of catheter pulmonary artery angiogram and pressure wave forms before dilation (c1) and catheter LPA angiogram after dilation (c2) at 10 months. The LPA was stenosed at the bifurcation (arrow). Balloon angioplasty improved the stenosis and significantly improved the pressure waveform. MPA = main pulmonary artery; RPA = right pulmonary artery; LPA = left pulmonary artery; DsAo = descending aorta.