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June 27, 2026Pediatric CardiologyOpen Access

Off-Label Sotatercept Use in Pediatric Pulmonary Arterial Hypertension: A Single-Center Retrospective Hemodynamic Study

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Why the study?

Sotatercept has shown efficacy in adults with PAH, but pediatric data remain limited to ongoing clinical trials.

Does off-label sotatercept improve hemodynamic parameters in pediatric patients with advanced pulmonary arterial hypertension refractory to triple therapy?

Comparison

Off-label sotatercept

Design

Single-center retrospective analysis

Follow-up

Approximately 24 weeks

Key result

Off-label sotatercept in 7 pediatric patients with advanced PAH refractory to triple therapy was associated with reductions in pulmonary vascular resistance index and mean pulmonary artery pressure.

Authors

DRDhruv RaoSHShylah HaldemanJCJeanne Carroll

Discussion

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Overview

Should not yet change practice in pediatric PAH; leaves open efficacy and safety pending prospective trials.

Key Points

  • The aim is to evaluate the hemodynamic impact of off-label sotatercept in pediatric patients with pulmonary arterial hypertension (PAH).
  • Single-center retrospective analysis of pediatric patients with PAH
  • All patients received maximal background triple therapy before sotatercept initiation
  • Right heart catheterization performed before and 24 weeks after treatment
  • Majority of patients showed reductions in pulmonary vascular resistance index (PVRI) and mean pulmonary artery pressure (mPAP)
  • Pulmonary capillary wedge pressure (PCWP) remained stable
  • Favorable hemodynamic trends observed at 24 weeks post-sotatercept initiation

Study Design

Type

Observational (n=7)

Multicenter

No

Structured PICO

Does off-label sotatercept improve hemodynamic parameters in pediatric patients with advanced pulmonary arterial hypertension refractory to triple therapy?

P
Population
7 pediatric patients (median age 14) with advanced WHO Group 1 or 3 PAH refractory to triple therapy, treated with off-label sotatercept and followed for approximately 24 weeks.
E
Exposure
Off-label sotatercept added to maximal background triple therapy.
O
Outcome
Hemodynamic parameters (including pulmonary vascular resistance index [PVRI], mean pulmonary artery pressure [mPAP], and pulmonary capillary wedge pressure [PCWP]) assessed via right heart catheterization at approximately 24 weeks.surrogate

Off-label sotatercept use in pediatric patients with advanced, refractory pulmonary arterial hypertension is associated with favorable hemodynamic improvements at 24 weeks, supporting the biologic plausibility for its use in this population.

Limitations

  • Small cohort
  • Highly selected pediatric cohort
  • Small sample size
  • Retrospective design

Cite This Study

Rao et al. (2026) conducted an observational in Pediatric Pulmonary Arterial Hypertension (n=7). Sotatercept was evaluated on Hemodynamic parameters including pulmonary vascular resistance index (PVRI), mean pulmonary artery pressure (mPAP), and pulmonary capillary wedge pressure (PCWP). Off-label sotatercept in 7 pediatric patients with advanced PAH refractory to triple therapy was associated with reductions in pulmonary vascular resistance index and mean pulmonary artery pressure.

synapsesocial.com/papers/6a3f6843aea7db3c1953f675https://doi.org/10.1007/s00246-026-04349-2
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