New England Journal of Medicine JULY 25, 2013

Riociguat for the Treatment of Chronic Thromboembolic Pulmonary Hypertension (CHEST-1)

Ghofrani HA, D'Armini AM, Grimminger F, et al.

Bottom Line

The CHEST-1 trial demonstrated that oral riociguat significantly improved exercise capacity and pulmonary hemodynamics in patients with inoperable or persistent/recurrent chronic thromboembolic pulmonary hypertension.

Key Findings

1. At 16 weeks, patients treated with riociguat experienced a significant improvement in the primary endpoint of 6-minute walk distance, with a least-squares mean difference of 46 meters compared to placebo (95% CI, 25 to 67; P<0.001).
2. Treatment with riociguat resulted in a significant reduction in pulmonary vascular resistance, with a least-squares mean difference of -246 dyn·sec·cm-5 compared to the placebo group (95% CI, -303 to -190; P<0.001).
3. Secondary endpoints, including N-terminal pro-B-type natriuretic peptide (NT-proBNP) levels (P<0.001) and World Health Organization (WHO) functional class (P=0.003), also showed statistically significant improvements in the riociguat arm.
4. The incidence of serious adverse events was comparable between the riociguat and placebo groups, with right ventricular failure (3% in both groups) and syncope being among the most common.

Study Design

Design
RCT
Double-Blind
Sample
261
Patients
Duration
16 wk
Median
Setting
Multicenter, international
Population Adults with inoperable chronic thromboembolic pulmonary hypertension or persistent/recurrent pulmonary hypertension following pulmonary endarterectomy.
Intervention Oral riociguat (dose-adjusted up to 2.5 mg three times daily).
Comparator Placebo.
Outcome Change from baseline in the distance walked in 6 minutes at week 16.

Study Limitations

The 16-week duration of the study is relatively short, limiting the ability to assess long-term clinical outcomes such as mortality or time to clinical worsening.
The study did not directly assess the impact of riociguat on right ventricular function or structure via imaging.
The trial was sponsored by the pharmaceutical manufacturer, which may introduce potential conflicts of interest.
Patients with severe renal or hepatic impairment were excluded, meaning findings may not be generalizable to these higher-risk subpopulations.

Clinical Significance

CHEST-1 established riociguat as the first evidence-based pharmacological therapy for patients with inoperable chronic thromboembolic pulmonary hypertension (CTEPH) or those with persistent/recurrent disease post-pulmonary endarterectomy, filling a critical gap in management for a condition previously lacking approved medical options.

Historical Context

Prior to the CHEST-1 trial, pulmonary endarterectomy (PEA) was the definitive gold-standard treatment for CTEPH, but many patients were deemed technically inoperable or continued to have pulmonary hypertension despite surgery. Before this trial, no pharmacologic agents had been approved specifically for this indication, and while other pulmonary hypertension medications were used off-label, their efficacy was not established by rigorous, large-scale randomized trials.

Guided Discussion

High-yield insights from every perspective

Med Student
Medical Student

What is the unique dual mechanism of action of riociguat, and why is this particularly advantageous in the pathophysiology of CTEPH compared to traditional PDE5 inhibitors like sildenafil?

Key Response

Riociguat is a soluble guanylate cyclase (sGC) stimulator. It has a dual mechanism: it increases the sensitivity of sGC to endogenous nitric oxide (NO) and also directly stimulates sGC independently of NO levels. In CTEPH, the pulmonary endothelium is often severely damaged, leading to low NO bioavailability. Unlike PDE5 inhibitors, which require endogenous NO to produce cGMP, riociguat can still generate cGMP and induce vasodilation even in NO-depleted environments.

Resident
Resident

In a patient diagnosed with CTEPH via V/Q scan and confirmed on right heart catheterization, what critical clinical step must be documented before initiating riociguat as per the CHEST-1 study protocol and current standards of care?

Key Response

Before starting riociguat, a patient must be evaluated by a multidisciplinary team (including a cardiothoracic surgeon) to determine if the disease is 'inoperable.' Pulmonary endarterectomy (PEA) remains the gold-standard treatment with curative potential. Riociguat is specifically indicated for those with inoperable CTEPH or those with persistent/recurrent pulmonary hypertension after surgical intervention.

Fellow
Fellow

While CHEST-1 met its primary endpoint of 6-minute walk distance (6MWD), it also showed a significant reduction in pulmonary vascular resistance (PVR). How should a fellow interpret a scenario where a patient's PVR improves significantly on riociguat but their 6MWD does not, and how does this reflect the limitations of 6MWD in the CTEPH population?

Key Response

6MWD is a global measure of exercise capacity influenced by age, musculoskeletal health, and deconditioning, which are common in chronic CTEPH. A fellow must recognize that PVR is a more direct measure of the pulmonary vascular response to the drug. A 'hemodynamic responder' who does not improve their 6MWD may require concurrent physical rehabilitation or evaluation for non-cardiac causes of exertional limitation rather than a change in PH-targeted therapy.

Attending
Attending

Reflecting on the serious adverse events in CHEST-1, particularly the incidence of hemoptysis, how does this finding impact your risk-benefit counseling and clinical monitoring for patients on riociguat therapy?

Key Response

CHEST-1 reported cases of serious hemoptysis, which is a known risk in CTEPH due to high-pressure bronchial artery collaterals and potential pulmonary infarction. Attendings must counsel patients that while riociguat is effective, any hemoptysis is a potential emergency. This necessitates a proactive monitoring strategy where new or worsening respiratory symptoms are evaluated with imaging to differentiate between drug side effects and disease-related vascular complications.

Scholarly Review

Critical appraisal through the lens of expert reviewers and guideline development

PhD
PhD

The CHEST-1 trial used a 16-week duration to assess its primary endpoint. From a clinical trial design perspective, what are the implications of using such a short timeframe for a chronic remodeling disease, and what alternative endpoints could better capture long-term right ventricular (RV) recovery?

Key Response

16 weeks is sufficient to capture acute hemodynamic vasodilation and functional improvement, but it may be too short to observe true reverse remodeling of the RV. As a PhD researcher, one would argue for long-term longitudinal studies using cardiac MRI (measuring RV ejection fraction and mass) or composite endpoints like 'time to clinical worsening' (TTCW), which provide a more robust assessment of disease progression and the durability of the treatment effect.

Journal Editor
Journal Editor

As a reviewer, how would you address the potential for 'unblinding' in the CHEST-1 trial caused by the mandatory dose-titration protocol based on systemic blood pressure?

Key Response

Because riociguat is titrated based on blood pressure (increasing the dose until a target is reached or hypotension occurs), investigators and patients might easily deduce who is in the treatment group versus the placebo group (where blood pressure remains stable). A rigorous editor would flag this as a threat to the internal validity of the subjective 6MWD results and would look for blinded 'central' assessors of the walk test to mitigate this expectation bias.

Guideline Committee
Guideline Committee

How does the evidence from CHEST-1 influence the strength of recommendation for riociguat in the ESC/ERS Guidelines for Pulmonary Hypertension, and should it be considered for off-label use in Group 1 PAH patients based on these results?

Key Response

CHEST-1 established riociguat as a Class I, Level B recommendation for inoperable or persistent CTEPH. However, guideline committees strictly separate this from Group 1 PAH (addressed in the PATENT-1 trial). A critical guideline point is the absolute contraindication of combining riociguat with PDE5 inhibitors due to severe hypotension risk—a recommendation grounded in the pharmacological understanding refined during these pivotal trials.

Clinical Landscape

Noteworthy Related Trials

2002

BREATHE-1 Trial

n = 213 · NEJM

Tested

Bosentan

Population

Patients with WHO functional class III or IV pulmonary arterial hypertension

Comparator

Placebo

Endpoint

Change in 6-minute walk distance

Key result: Bosentan improved exercise capacity and delayed clinical worsening in patients with severe pulmonary arterial hypertension.
2005

SUPER-1 Trial

n = 278 · NEJM

Tested

Sildenafil

Population

Symptomatic patients with pulmonary arterial hypertension

Comparator

Placebo

Endpoint

Change in 6-minute walk distance

Key result: Oral sildenafil significantly improved exercise capacity and hemodynamics in patients with pulmonary arterial hypertension.
2015

CHEST-2 Trial

n = 237 · Eur Respir J

Tested

Riociguat

Population

Patients with chronic thromboembolic pulmonary hypertension who completed CHEST-1

Comparator

None (Open-label extension)

Endpoint

Safety and long-term efficacy

Key result: Long-term riociguat therapy demonstrated sustained improvements in exercise capacity and was well-tolerated.

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