Key result
Veins without baseline distal disease linked to ~269% higher odds of minimal disease in pediatric PVS.
Why the study?
Patient and pulmonary vein characteristics predictive of individual vein outcomes in pediatric intraluminal pulmonary vein stenosis were not well defined.
What patient and pulmonary vein characteristics predict individual vein outcomes in pediatric pulmonary vein stenosis treated with imatinib mesylate +/- bevacizumab?
Cohort (n=46)
Open-label
No
What patient and pulmonary vein characteristics predict individual vein outcomes in pediatric pulmonary vein stenosis treated with imatinib mesylate +/- bevacizumab?
In pediatric patients with multi-vessel pulmonary vein stenosis treated with imatinib mesylate +/- bevacizumab, absence of distal disease, non-left upper vein location, and age ≥ 1 year predict better individual vessel patency at 72 weeks.
May support baseline risk stratification in pediatric multi-vessel PVS; hypothesis-generating and requires prospective validation before guiding care.
The purpose of this study was to determine what patient and pulmonary vein characteristics at the diagnosis of intraluminal pulmonary vein stenosis (PVS) are predictive of individual vein outcomes. A retrospective, single-center, cohort sub-analysis of individual pulmonary veins of patients enrolled in the clinical trial NCT00891527 using imatinib mesylate +/− bevacizumab as adjunct therapy for the treatment of multi-vessel pediatric PVS between March 2009 and December 2014 was performed. The 72-week outcomes of the individual veins are reported. Among the 48 enrolled patients, 46 patients and 182 pulmonary veins were included in the study. Multivariable analysis demonstrated that patients with veins without distal disease at baseline (odds ratio, OR 3.69, 95% confidence interval, CI [1.52, 8.94], p = 0.004), location other than left upper vein (OR 2.58, 95% CI [1.07, 6.19], p = 0.034), or veins in patients ≥ 1 y/o (OR 5.59, 95% CI [1.81, 17.3], p = 0.003) were at higher odds of having minimal disease at the end of the study. Veins in patients who received a higher percentage of eligible drug doses required fewer reinterventions (IRR 0.76, 95% CI [0.68, 0.85], p < 0.001). The success of a multi-modal treatment approach to aggressive PVS depends on the vein location, disease severity, and drug dose intensity.
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Callahan et al. (2021) conducted a cohort in Pediatric intraluminal pulmonary vein stenosis (PVS) (n=46). No distal disease at baseline vs. Distal disease at baseline was evaluated on Minimal disease at 72 weeks (OR 3.69, 95% CI 1.52-8.94, p=0.004). Veins without distal disease at baseline were associated with significantly higher odds of having minimal disease at 72 weeks (OR 3.69) in pediatric patients with multi-vessel pulmonary vein stenosis.
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