The Lancet Neurology MAY 15, 2017

Safety and efficacy of edaravone in well defined patients with amyotrophic lateral sclerosis: a randomised, double-blind, placebo-controlled trial

Writing Group; Edaravone ALS Study Group (Abe K et al.)

Bottom Line

In a prospectively defined subpopulation of early-stage, rapidly progressing ALS patients, intravenous edaravone significantly slowed the decline of physical function compared to placebo over 24 weeks.

Key Findings

1. Among the 137 randomized patients, the least-squares mean change in the ALSFRS-R score at 24 weeks was -5.01 (SE 0.64) in the edaravone group versus -7.50 (SE 0.66) in the placebo group.
2. Edaravone treatment resulted in a statistically significant reduction in functional decline, with a treatment difference of 2.49 points (95% CI, 0.99 to 3.98; p=0.0013) compared to placebo.
3. The incidence of adverse events was similar between the two groups, with serious adverse events reported in 16% (11 of 68) of patients receiving edaravone and 16% (11 of 69) of patients receiving placebo.

Study Design

Design
RCT
Double-Blind
Sample
137
Patients
Duration
24 wk
Median
Setting
31 hospitals, Japan
Population Adults aged 20-75 with definite/probable ALS (El Escorial criteria), disease duration ≤ 2 years, normal respiratory function (FVC ≥ 80%), and a 1-4 point decline in ALSFRS-R during a 12-week observational run-in phase.
Intervention Intravenous edaravone 60 mg daily (administered for 14 days in cycle 1, then 10 of 14 days in subsequent cycles, followed by a 14-day drug-free period per 28-day cycle).
Comparator Intravenous placebo (saline) administered on an identical 28-day cycle schedule.
Outcome Change in the Revised ALS Functional Rating Scale (ALSFRS-R) score from baseline to 24 weeks.

Study Limitations

The 24-week follow-up period was relatively short, preventing definitive conclusions regarding edaravone's impact on long-term overall survival or time to mechanical ventilation.
The highly restrictive inclusion criteria meant that only a small, specific subset of early-stage ALS patients (estimated at around 7% of the general ALS population) were studied, limiting generalizability to those with more advanced disease.
The tested regimen required burdensome, frequent intravenous infusions, though this limitation was later addressed by the development of an approved oral formulation.

Clinical Significance

This pivotal trial directly led to the 2017 FDA approval of edaravone (Radicava), ending a 22-year drought of new ALS therapies in the United States. It also demonstrated the profound value of selective patient enrichment in neurodegenerative clinical trials.

Historical Context

For over two decades following the 1995 approval of riluzole, ALS drug development saw a string of high-profile Phase 3 trial failures—most notably the EMPOWER trial of dexpramipexole in 2013 (which likely prompted the query's conflation of EMPOWER and edaravone). Edaravone itself initially failed its first broad Phase 3 trial (Study 16). However, a post-hoc analysis suggested benefit in early-stage, rapid progressors. By prospectively enriching the Study 19 trial population based on these strict criteria, investigators successfully demonstrated efficacy, marking a paradigm shift in ALS trial design.

Guided Discussion

High-yield insights from every perspective

Med Student
Medical Student

What is the proposed mechanism of action of edaravone in amyotrophic lateral sclerosis, and how does it target the underlying pathophysiology of the disease?

Key Response

Edaravone is a free radical scavenger. Oxidative stress is hypothesized to play a major role in motor neuron degeneration in ALS. Understanding this mechanism helps link basic science to the drug's intended neuroprotective effect, though it is not a curative treatment.

Resident
Resident

Based on the specific inclusion criteria of this trial, which subset of ALS patients is most likely to benefit from edaravone, and what are the practical logistical challenges of its intravenous administration?

Key Response

The trial specifically enrolled patients with early-stage disease, good respiratory function, independent living, and documented rapid progression. Furthermore, intravenous edaravone requires cyclic daily infusions, creating a major logistical burden that heavily impacts outpatient management decisions.

Fellow
Fellow

The primary endpoint of the trial was the change in the ALSFRS-R score. What are the limitations of the ALSFRS-R in detecting meaningful clinical change, and how should a 2.49-point difference between groups over 24 weeks be interpreted in practice?

Key Response

The ALSFRS-R is a non-linear ordinal scale where a single point change can represent vastly different functional losses depending on the domain affected. A 2.49-point difference is statistically significant, but its true clinical meaningfulness is highly debated among neuromuscular specialists.

Attending
Attending

Given that the trial's success relied on a highly enriched patient population, how do you navigate shared decision-making regarding edaravone initiation for patients who fall outside these strict criteria?

Key Response

Attendings must balance the trial's narrow evidence base with the desperation of ALS patients. Prescribing it broadly involves discussing the lack of proven benefit in advanced disease, financial toxicity, and side effects versus the patient's strong desire for any disease-modifying therapy.

Scholarly Review

Critical appraisal through the lens of expert reviewers and guideline development

PhD
PhD

This trial succeeded after a broader Phase 3 trial failed, leading researchers to use a post-hoc defined subpopulation for this prospective study. What are the statistical and methodological implications of using post-hoc enrichment strategies for a confirmatory trial?

Key Response

While prospectively testing a post-hoc hypothesis is valid, it significantly restricts generalizability and raises questions about whether the effect is a true biological interaction dependent on disease stage or a potential artifact of subgroup data dredging from the previous failed trial.

Journal Editor
Journal Editor

The trial was conducted entirely in a single country with a follow-up period of only 24 weeks. What major concerns regarding external validity and trial duration should be flagged during peer review?

Key Response

A 24-week trial for a progressive disease with a 3 to 5 year lifespan provides limited data on long-term survival or delayed need for mechanical ventilation. Furthermore, a homogenous cohort raises questions about global applicability, pharmacogenomics, and differences in standard of care.

Guideline Committee
Guideline Committee

How should ALS management guidelines grade the evidence for edaravone considering the conflict between broad regulatory approvals and the trial's narrow inclusion criteria, and how does this compare to established recommendations for riluzole?

Key Response

Edaravone has restricted evidence for slowing functional decline in a specific early-stage subset, unlike riluzole which has established Level A evidence for prolonging survival. Committees must decide whether to restrict recommendations to the trial's strict criteria, which directly impacts payer coverage and global clinical pathways.

Clinical Landscape

Noteworthy Related Trials

1994

Riluzole ALS Trial

n = 155 · NEJM

Tested

Riluzole 100 mg daily

Population

Patients with amyotrophic lateral sclerosis (ALS)

Comparator

Placebo

Endpoint

Survival (time to death or tracheostomy)

Key result: Riluzole significantly improved survival in patients with ALS, establishing it as the first approved disease-modifying therapy for the condition.
2013

EMPOWER Trial

n = 943 · Lancet Neurol

Tested

Dexpramipexole 150 mg twice daily

Population

Patients with familial or sporadic ALS

Comparator

Placebo

Endpoint

Combined assessment of function and survival (CAFS)

Key result: Dexpramipexole showed no significant efficacy over placebo in slowing functional decline or improving survival in a broad ALS population.
2020

CENTAUR Trial

n = 137 · NEJM

Tested

Sodium phenylbutyrate-taurursodiol (AMX0035)

Population

Patients with definite ALS and symptom onset within 18 months

Comparator

Placebo

Endpoint

Rate of decline in the ALSFRS-R score

Key result: AMX0035 resulted in a significantly slower functional decline than placebo over a 24-week period.

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