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Extrinsic Compression Of Bilateral Upper Pulmonary Veins By Pulmonary Artery Flow Restrictors
Calvin MacDonald, BS1, Yalim Yalcin, MD2, Katherine Braley, MD2, Camilla Rossi, MD2, Jennifer Nelson, MD, MS2.
1University of Central Florida College of Medicine, Orlando, FL, USA, 2Nemours Children's Hospital, Orlando, FL, USA.
Objective: To describe pulmonary vein stenosis related to the implantation of pulmonary flow restrictors (PFRs) in an infant with congenital heart disease.
Methods: A premature infant with trisomy 21 presented with complete atrioventricular septal defect (AVSD). Initial palliation with PFRs was performed given significant comorbid conditions and low birth weight. Bilateral PFRs remained in place for four months.
Results: While awaiting repair, the patient underwent surveillance echocardiograms noting bilateral upper pulmonary vein stenosis. At 6 months, surgical repair was planned, including repair of pulmonary vein stenosis. As preoperative CTA suggested compression of the upper pulmonary veins by the PFR devices (
Figure), the surgical plan was modified for removal of the PFRs first, with re-evaluation of pulmonary veins intraoperatively, prior to pulmonary vein intervention. Removing the flow restrictors relieved the compression. Concomitant AVSD repair was performed with an uneventful recovery.
Conclusions: Percutaneous PFRs are an alternative to pulmonary artery banding, but may cause extraluminal pulmonary vein compression, mimicking intrinsic stenosis. Cross-sectional imaging and heightened awareness of this complication in the differential diagnosis are essential when planning surgical treatment for patients with PFRs. When extrinsic compression is detected, removing the PFRs can offer relief of gradients, without the need for pulmonary vein reconstruction. This report underscores the importance of an expanded differential diagnosis for pulmonary vein stenosis in the context of increasing utilization of implanted bilateral PFRs.
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