ArticleJACC. Advances2024
Systemic Sirolimus Therapy Is Associated With Reduced Intervention Frequency in Pulmonary Vein Stenosis.
Article in JACC. Advances, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
10 citing papers in PubMed.
- Imatinib Mesylate and Sirolimus Inhibit Vascular Fibroproliferative Remodeling in Pulmonary Vein Stenosis.Children (Basel, Switzerland) · 2026Article
- A translational framework for patient-specific modeling of pulmonary vein stenosis.JTCVS open · 2026Article
- Systemic Sirolimus Monitoring in Pediatric Pulmonary Vein Stenosis.Pediatric cardiology · 2026Article
- Therapeutic Targets for Pediatric Pulmonary Vein Stenosis: Insights from Animal Models.Children (Basel, Switzerland) · 2026Review
- Characteristics and outcomes of preterm infants with pulmonary vein stenosis in the contemporary era: a PHIS database analysis.Journal of perinatology : official journal of the California Perinatal Association · 2026Article
- Catheter and echocardiographic-focused management of primary pulmonary vein stenosis in premature infants: a single center 5-year experience.Journal of perinatology : official journal of the California Perinatal Association · 2026Article
- Case Report: Post-Total anomalous pulmonary venous connection pulmonary hypertension - novel treatment using sirolimus and atrial flow regulator implantation.Frontiers in cardiovascular medicine · 2026Article
- Transcatheter Management of Pulmonary Vein Stenosis in Children.Pediatric cardiology · 2025Review
- Evaluation of Treatment Effect of Sirolimus on Pediatric Pulmonary Vein Stenosis Using a Neonatal Rat Model Induced by Bilateral Pulmonary Vein Banding.JACC. Basic to translational science · 2025Article
- Serial echocardiography in preterm infants with bronchopulmonary dysplasia: diagnosing and managing recurrent pulmonary vein stenosis.Case reports in perinatal medicine · 2025Article
Corrections and comments
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Authors and funding
18 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Background: Early clinical outcomes data for adjunctive systemic sirolimus therapy (SST) for moderate to severe pediatric pulmonary vein stenosis (PVS) are promising but limited. Objectives: The authors aimed to characterize a cohort of patients treated with SST to determine if SST was associated with a reduction in frequency of PVS interventions. Methods: Medical records of 45 patients with PVS treated with SST for ≥1 month from 2015 to 2022 were retrospectively reviewed. PVS intervention rates pre-SST and on-SST were compared using generalized Poisson mixed models, accounting for paired intervals within each patient. In addition to an unadjusted model, an adjusted model accounted for age at interval start, PVS type, sex, prematurity, and concurrent antiproliferative therapy. Mean number of PVS interventions per patient over time (mean cumulative function) were also compared for these intervals in an unpaired fashion. Kaplan-Meier estimates were used to quantify survival over time. Results: Median per-patient PVS intervention rate (interventions/year) was 5 pre-SST and 1.7 on-SST, significantly lower on-SST in the unadjusted and adjusted models ( Conclusions: SST was associated with a reduction in frequency of PVS interventions. Prospective studies are warranted to determine potential causality, delineate patient- and vein-level outcomes, and determine optimal therapeutic duration.
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Registered trials
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