New England Journal of Medicine APRIL 20, 2023

Phase 3 Trial of Sotatercept for Treatment of Pulmonary Arterial Hypertension

Marius M. Hoeper, David B. Badesch, Hossein-Ardeschir Ghofrani, et al.

Bottom Line

In patients with symptomatic pulmonary arterial hypertension on stable background therapy, the addition of subcutaneous sotatercept significantly improved exercise capacity, as measured by the 6-minute walk distance, and reduced the risk of clinical worsening or death.

Key Findings

1. The primary endpoint was met, with a mean placebo-adjusted increase in 6-minute walk distance (6MWD) of 40.8 meters (95% CI, 27.5 to 54.1; P<0.001) in the sotatercept group at 24 weeks.
2. Sotatercept demonstrated an 84% reduction in the hazard ratio for the occurrence of death or clinical worsening events compared to placebo (HR, 0.16; 95% CI, 0.08 to 0.35; P<0.001).
3. Secondary outcomes were largely favorable, including significant improvements in pulmonary vascular resistance (PVR), N-terminal pro-B-type natriuretic peptide (NT-proBNP) levels, and World Health Organization (WHO) functional class.
4. Common adverse events associated with sotatercept included telangiectasia (10.4% vs. 0% in placebo), epistaxis (21.5% vs. 3.1%), and increased hemoglobin levels.

Study Design

Design
RCT
Double-Blind
Sample
323
Patients
Duration
24 wk
Median
Setting
Multicenter, International
Population Adults with WHO Group 1 pulmonary arterial hypertension (functional class II or III) receiving stable background PAH therapy.
Intervention Subcutaneous sotatercept (0.3 mg/kg starting dose, target 0.7 mg/kg) every 3 weeks.
Comparator Subcutaneous matching placebo every 3 weeks.
Outcome Change from baseline at week 24 in the 6-minute walk distance (6MWD).

Study Limitations

The trial duration of 24 weeks is relatively short for assessing long-term durability and the full safety profile of this novel therapeutic.
The trial excluded patients with very severe functional impairment (WHO functional class IV) and those with specific comorbidities, potentially limiting generalizability to the most advanced or complex PAH populations.
The frequency of specific side effects such as telangiectasia and epistaxis may have compromised the blinding of investigators and patients to the treatment allocation.
The study was conducted in patients on stable background therapy, meaning the results may not be directly applicable to treatment-naive patients or those not yet on standardized PAH therapy.

Clinical Significance

Sotatercept represents a first-in-class activin signaling inhibitor that shifts the treatment paradigm from simple vasodilation to targeting the underlying vascular remodeling in PAH. These results support its use as an add-on therapy for patients with symptomatic PAH who remain symptomatic despite standard-of-care background treatment.

Historical Context

For decades, the pharmacologic management of pulmonary arterial hypertension has been dominated by pulmonary vasodilators (endothelin receptor antagonists, phosphodiesterase-5 inhibitors, and prostacyclin pathway agonists) which primarily focus on hemodynamic improvement. Sotatercept, by modulating the imbalance between pro-proliferative and anti-proliferative signaling pathways (specifically the activin/TGF-β pathway), introduces a novel mechanism of action—vascular reverse remodeling—that addresses the pathophysiology of PAH rather than just the symptoms.

Guided Discussion

High-yield insights from every perspective

Med Student
Medical Student

How does the mechanism of action of sotatercept fundamentally differ from traditional pulmonary arterial hypertension (PAH) therapies such as endothelin receptor antagonists (ERAs) or phosphodiesterase-5 (PDE5) inhibitors?

Key Response

Conventional PAH therapies primarily target three pathways—endothelin, nitric oxide, and prostacyclin—to induce vasodilation of the pulmonary vasculature. In contrast, sotatercept is a first-in-class activin signaling inhibitor (a fusion protein) that acts as a ligand trap for activins and growth differentiation factors. By sequestering these ligands, it rebalances the signaling between the pro-proliferative TGF-̢̢ superfamily pathways and the anti-proliferative BMPR2 pathway, theoretically reversing the vascular remodeling rather than just inducing acute vasodilation.

Resident
Resident

Based on the adverse event profile identified in the STELLAR trial, what specific laboratory and physical examination monitoring parameters are required for a patient starting sotatercept?

Key Response

The STELLAR trial identified specific safety signals associated with sotatercept's mechanism. Residents must monitor for erythrocytosis (increased hemoglobin levels) and thrombocytopenia, necessitating regular CBC monitoring. Additionally, physical exams should focus on the development of telangiectasias (observed in 14.1% of the sotatercept group) and signs of epistaxis, as these were significantly more common in the treatment arm compared to placebo.

Fellow
Fellow

In the STELLAR trial, 60% of patients were on background triple therapy (including a parenteral prostacyclin). Does the magnitude of the reduction in pulmonary vascular resistance (PVR) seen with sotatercept suggest a ceiling effect for current therapies, and how does this affect our hemodynamic targets in advanced PAH?

Key Response

Sotatercept achieved a median reduction in PVR of 234.6 dyn"sec"cm⁻⁵ even in patients already optimized on therapy. This suggests that the remodeling-based pathology of PAH is not fully addressed by maximal vasodilator therapy. For fellows, this highlights that hemodynamic 'normalization' might become a more realistic goal, moving beyond simply improving functional class toward addressing the structural drivers of right heart afterload.

Attending
Attending

The STELLAR trial demonstrated a significant 40.8-meter improvement in 6-minute walk distance (6MWD). How should we weigh this functional improvement against the long-term uncertainty of telangiectasias and potential vascular malformations in a young PAH population?

Key Response

As a practice-changing trial, the 6MWD improvement is clinically meaningful, but the development of telangiectasias raises questions about off-target systemic effects on angiogenesis. For an attending, the decision to prescribe involves balancing the immediate reduction in clinical worsening (84% lower risk) with the unknown long-term sequelae of chronic activin inhibition, especially in patients who may remain on this therapy for decades.

Scholarly Review

Critical appraisal through the lens of expert reviewers and guideline development

PhD
PhD

The primary endpoint analysis utilized a rank-based ANCOVA to account for missing data due to death or clinical worsening. How does this specific statistical choice protect against survivor bias compared to a standard Last Observation Carried Forward (LOCF) approach?

Key Response

In PAH trials, patients who die cannot complete the 6MWD. Using a rank-based ANCOVA allows investigators to assign the 'worst possible rank' to those who died or experienced clinical worsening. This is more robust than LOCF, which might inappropriately maintain a 'good' baseline walk distance for a patient who later died, thereby ensuring that the efficacy results reflect a true clinical benefit that accounts for attrition due to disease progression.

Journal Editor
Journal Editor

While the STELLAR trial is undeniably positive, the 24-week duration is relatively brief for a life-long disease. To what extent does the surrogate endpoint of 6MWD correlate with long-term survival in this specific drug class, and should the FDA have required a longer morbidity/mortality-driven primary endpoint?

Key Response

Editors look for the 'vulnerability' in a study. While 6MWD is the standard regulatory endpoint, its correlation with long-term survival has been questioned in recent years. Given that sotatercept is a first-in-class biologic with systemic vascular effects, a reviewer would flag the need for long-term safety data (extension studies) to ensure that the early hemodynamic and functional gains translate into sustained survival without prohibitive late-onset toxicity.

Guideline Committee
Guideline Committee

Given the results of the STELLAR trial, should sotatercept be integrated into the ESC/ERS PAH treatment algorithm as a standard add-on for patients in intermediate-risk categories, or should it be reserved for those failing triple therapy?

Key Response

The 2022 ESC/ERS guidelines currently advocate for a risk-stratified approach, often leading to triple therapy for higher-risk patients. Because sotatercept showed benefit across background therapy levels (dual and triple), the committee must decide if it merits a Class I recommendation for all Group 1 PAH patients with functional limitations, potentially displacing or augmenting the role of parenteral prostacyclins which carry significantly higher treatment burdens and complications.

Clinical Landscape

Noteworthy Related Trials

2013

SERAPHIN Trial

n = 742 · NEJM

Tested

Macitentan

Population

Symptomatic pulmonary arterial hypertension

Comparator

Placebo

Endpoint

Time to first morbidity or mortality event

Key result: Macitentan significantly reduced the risk of morbidity and mortality in patients with PAH.
2015

AMBITION Trial

n = 500 · NEJM

Tested

Ambrisentan plus Tadalafil

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Treatment-naive pulmonary arterial hypertension patients

Comparator

Ambrisentan or Tadalafil monotherapy

Endpoint

Time to first clinical failure event

Key result: Initial combination therapy significantly reduced the risk of clinical failure compared with individual monotherapies.
2015

GRIPHON Trial

n = 1,156 · NEJM

Tested

Selexipag

Population

Symptomatic pulmonary arterial hypertension

Comparator

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Endpoint

Composite of morbidity and mortality

Key result: Selexipag reduced the risk of a morbidity or mortality event by 40 percent compared to placebo.

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