Journal of the American College of Cardiology July 12, 2022

Myosin Inhibition in Patients With Obstructive Hypertrophic Cardiomyopathy Referred for Septal Reduction Therapy

Milind Y. Desai, Anjali Owens, Jeffrey B. Geske, et al.

Bottom Line

In patients with severely symptomatic obstructive hypertrophic cardiomyopathy eligible for septal reduction therapy, the addition of mavacamten significantly reduced the need for invasive procedures and improved symptoms compared to placebo.

Key Findings

1. At 16 weeks, 17.9% (10 of 56) of patients in the mavacamten group met the primary composite endpoint of proceeding with or remaining eligible for septal reduction therapy (SRT), compared to 76.8% (43 of 56) in the placebo group.
2. The absolute treatment difference was 58.9% in favor of mavacamten (95% CI, 44.0% to 73.9%; P < 0.001).
3. Mavacamten significantly improved NYHA functional class, with 63% of treated patients experiencing a ≥1 class improvement compared to 21% in the placebo group.
4. Patients receiving mavacamten demonstrated significant reductions in resting and provoked left ventricular outflow tract (LVOT) gradients, alongside meaningful improvements in the Kansas City Cardiomyopathy Questionnaire Clinical Summary Score (KCCQ-CSS).

Study Design

Design
Randomized Clinical Trial
Double-Blind
Sample
112
Patients
Duration
16 weeks
Median
Setting
Multicenter, US
Population Adults with symptomatic obstructive hypertrophic cardiomyopathy (NYHA class III-IV or class II with exertional syncope) who remained severely symptomatic despite maximally tolerated medical therapy and were actively referred for septal reduction therapy.
Intervention Mavacamten (started at 5 mg daily, titrated up to 15 mg based on echocardiographic parameters), added to maximally tolerated background medical therapy.
Comparator Placebo added to maximally tolerated background medical therapy.
Outcome A composite endpoint of the patient's decision to proceed with septal reduction therapy (SRT) or continuing to meet the 2011 ACC/AHA guideline criteria for SRT eligibility at 16 weeks.

Study Limitations

The primary endpoint was evaluated at a short follow-up duration of 16 weeks, limiting the initial assessment of long-term safety and durability of SRT avoidance (though long-term extension data were subsequently collected).
The rigorous echocardiographic monitoring required for safe dose titration to prevent left ventricular systolic dysfunction may pose logistical challenges in routine real-world clinical practice.
The relatively small sample size (N=112) limits the statistical power to detect rare but serious adverse events, such as profound drops in left ventricular ejection fraction.
The highly selected patient population (those already referred for SRT at specialized expert centers) may affect generalizability to broader, less symptomatic obstructive HCM populations.

Clinical Significance

VALOR-HCM demonstrated that the first-in-class cardiac myosin inhibitor mavacamten provides a highly effective medical alternative to invasive septal reduction therapy (SRT) for severely symptomatic obstructive HCM patients. By directly mitigating the underlying sarcomeric hypercontractility, mavacamten significantly relieves left ventricular outflow tract obstruction and improves functional status, fundamentally shifting the treatment paradigm from broad symptom management and invasive procedures toward targeted pharmacological disease modification.

Historical Context

For decades, medical therapy for obstructive HCM relied on non-disease-specific agents like beta-blockers, non-dihydropyridine calcium channel blockers, and disopyramide, which often failed to sufficiently control symptoms. Patients with refractory symptoms were typically referred for surgical myectomy or alcohol septal ablation, which are highly effective but carry inherent periprocedural risks and require specialized institutional expertise. Following the EXPLORER-HCM trial (2020), which validated mavacamten's efficacy in improving general exercise capacity, VALOR-HCM was designed to specifically evaluate its utility in a sicker, SRT-eligible cohort. Its success established mavacamten as a viable alternative to invasive therapies, marking a landmark advancement in precision medicine for HCM.

Guided Discussion

High-yield insights from every perspective

Med Student
Medical Student

How does the mechanism of action of mavacamten specifically target the underlying pathophysiology of obstructive hypertrophic cardiomyopathy (oHCM) differently than traditional first-line therapies like beta-blockers?

Key Response

Mavacamten is a first-in-class cardiac myosin inhibitor that directly reduces the number of actin-myosin cross-bridges. This specifically targets the hypercontractility and impaired relaxation characteristic of oHCM at the sarcomere level. In contrast, traditional therapies like beta-blockers decrease inotropy and chronotropy indirectly through adrenergic blockade, rather than altering the fundamental molecular defect of the sarcomere.

Resident
Resident

A patient with severely symptomatic oHCM is started on mavacamten based on the VALOR-HCM trial to avoid septal reduction therapy. What is the most critical safety parameter that must be serially monitored, and how might this influence the choice between medical and procedural management?

Key Response

Left ventricular ejection fraction (LVEF) must be rigorously monitored via a Risk Evaluation and Mitigation Strategy (REMS) program because mavacamten's negative inotropy can induce systolic heart failure. If LVEF drops below 50%, the drug must be interrupted. Clinicians must weigh the burden of serial echocardiography and risk of heart failure against the upfront risks of invasive septal reduction therapy.

Fellow
Fellow

While VALOR-HCM demonstrated a robust reduction in the 16-week eligibility for septal reduction therapy (SRT), how should you counsel a young, healthy 35-year-old oHCM patient regarding the choice between a novel pharmacologic agent and surgical myectomy at a comprehensive HCM center?

Key Response

Counseling requires complex shared decision-making. Mavacamten offers a non-invasive medical alternative but necessitates a lifelong commitment to the drug, strict REMS monitoring, and carries long-term unknown risks. Conversely, surgical myectomy at a high-volume center of excellence provides a permanent, definitive cure for the outflow gradient with excellent long-term survival and extremely low complication rates, potentially freeing the patient from lifelong medical therapy.

Attending
Attending

Given the profound success of mavacamten in reducing the need for septal reduction therapy (SRT) in VALOR-HCM, how might the widespread clinical adoption of this drug paradoxically impact the outcomes of patients who ultimately fail medical therapy and require surgical intervention?

Key Response

Septal myectomy and alcohol septal ablation are highly specialized procedures where operator volume is inversely correlated with mortality and complications. If mavacamten drastically reduces the number of patients referred for SRT, comprehensive HCM centers may experience a drop in procedural volume, potentially degrading surgical skill maintenance and worsening outcomes for the subset of patients who eventually require invasive intervention.

Scholarly Review

Critical appraisal through the lens of expert reviewers and guideline development

PhD
PhD

VALOR-HCM utilized a primary composite endpoint defined as the patient's decision to proceed with SRT or still meeting guideline criteria for SRT at 16 weeks. What are the methodological vulnerabilities of combining a subjective decision with objective criteria in a short-term trial for a chronic condition?

Key Response

A 16-week window is methodologically brief for a lifelong genetic condition, potentially missing late adverse effects or tachyphylaxis. Furthermore, the subjective component (patient decision) can be heavily influenced by the trial environment; patients experiencing the intense monitoring burden of the trial might alter their perception of surgical risk versus medical management, introducing ascertainment bias into the composite primary endpoint.

Journal Editor
Journal Editor

As a peer reviewer evaluating the VALOR-HCM manuscript, how would you address the potential for functional unblinding, and how might this impact the validity of the subjective components of the primary endpoint?

Key Response

Because mavacamten predictably and rapidly reduces left ventricular outflow tract gradients and LVEF, both the treating clinicians and the patients (who undergo serial safety echocardiograms) could likely deduce their treatment allocation. A rigorous reviewer would flag this functional unblinding as a major threat to validity, particularly because one half of the primary endpoint relies on the patient's subjective decision to proceed with or defer surgery.

Guideline Committee
Guideline Committee

Current AHA/ACC guidelines list septal reduction therapy (SRT) as a Class 1 recommendation for severely symptomatic oHCM refractory to maximum tolerated medical therapy. Based on VALOR-HCM, should mavacamten be elevated to a prerequisite Class 1 recommendation prior to SRT, and what evidence gaps remain?

Key Response

VALOR-HCM provides Level of Evidence B-R that mavacamten significantly reduces SRT eligibility in refractory patients. Guidelines must consider positioning cardiac myosin inhibitors as a mandatory intermediate step (Class 1 or 2a) between standard non-vasodilating therapies and invasive SRT. However, the committee must balance this against the lack of multi-decade safety data for mavacamten compared to the well-established long-term survival data following surgical myectomy.

Clinical Landscape

Noteworthy Related Trials

2018

PIONEER-HCM Trial

n = 21 · Ann Intern Med

Tested

Mavacamten

Population

Symptomatic obstructive hypertrophic cardiomyopathy (oHCM)

Comparator

No comparator (open-label, dose-finding)

Endpoint

Change in postexercise LVOT gradient at 12 weeks

Key result: Mavacamten treatment eliminated LVOT obstruction in most patients and was associated with improvements in symptoms and exercise capacity.
2020

EXPLORER-HCM Trial

n = 251 · Lancet

Tested

Mavacamten (2.5 to 15 mg daily)

Population

Symptomatic obstructive hypertrophic cardiomyopathy (oHCM)

Comparator

Placebo

Endpoint

Composite of pVO2 increase and NYHA class improvement

Key result: Mavacamten significantly improved exercise capacity, LVOT obstruction, NYHA functional class, and health status.
2024

SEQUOIA-HCM Trial

n = 282 · NEJM

Tested

Aficamten (5 to 20 mg daily)

Population

Symptomatic obstructive hypertrophic cardiomyopathy (oHCM)

Comparator

Placebo

Endpoint

Change in peak oxygen uptake (pVO2) at 24 weeks

Key result: Aficamten significantly improved exercise capacity, symptoms, and reduced LVOT gradients with a favorable safety profile.

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