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HEALEY ALS Platform Trial

Operational Development and Launch of an Adaptive Platform Trial in Amyotrophic Lateral Sclerosis: Processes and Learnings From the First Four Regimens

Year of Publication: 2025

Authors: Harkey BA, Distefano S, Pagliaro JA, ..., Sherman AV

Journal: Muscle & Nerve

Citation: Muscle Nerve. 2025;72(2):294-305

Link: https://onlinelibrary.wiley.com/doi/10.1002/mus.28442


Clinical Question

Can an adaptive platform trial design provide operational advantages for testing multiple ALS therapeutics concurrently?

Bottom Line

The HEALEY ALS Platform Trial demonstrated that an adaptive platform design enables concurrent testing of multiple ALS treatments with shared placebo and accelerated regulatory timelines. While none of the first four regimens showed efficacy, the infrastructure proved operationally efficient.

Major Points

  • Master protocol achieved single IRB approval in 120 days; fourth regimen amendment approved in just 21 days -- demonstrating regulatory efficiency of platform design
  • Enrollment completed in 15 months for Regimens A-C and 11 months for Regimen D; 84-90% completion rates
  • 54 NEALS consortium sites participated across the US with concurrent enrollment into multiple regimens
  • Shared placebo pool of 122-164 participants increased statistical power while reducing total placebo exposure
  • None of five tested regimens met efficacy threshold: Pridopidine DRR 0.99 (95% CrI 0.80-1.21), Verdiperstat DRR 0.98 (0.77-1.24), CNM-Au8 DRR 0.97 (0.78-1.18), Zilucoplan DRR 1.08, Trehalose DRR 0.87
  • CNM-Au8 showed significant biomarker effect (plasma NfL decreased 2.3% vs increased 7.9% in placebo, p=0.04) but no clinical benefit
  • Individual regimen results published separately: Pridopidine in JAMA 2025, Verdiperstat in JAMA Neurol 2025, CNM-Au8 in JAMA 2025

Design

Study Type: Adaptive platform trial, phase 2/3, multicenter, randomized, double-blind, placebo-controlled

Randomization: 1

Blinding: Double-blind

Enrollment Period: August 2020 - February 2023

Follow-up Duration: 24 weeks per regimen

Centers: 54

Countries: United States

Sample Size: 653

Analysis: Bayesian shared-parameter joint model; disease rate ratio (DRR); 3:1 randomization with shared placebo


Inclusion Criteria

  • Adults with ALS (clinically possible, probable, or definite per revised El Escorial criteria)
  • Disease duration 36 months or less
  • Pulmonary vital capacity (SVC) 50% or greater of predicted
  • Ability to swallow pills and liquids
  • Stable dosing (>=30 days) or no prior use of riluzole and/or edaravone
  • QTcF interval <450 ms (males) or <470 ms (females)
  • Stratification by concurrent edaravone and/or riluzole use

Exclusion Criteria

  • Disease duration >36 months
  • SVC <50% predicted
  • Inability to swallow oral medications
  • Active participation in another interventional clinical trial

Arms

FieldRegimen A - ZilucoplanRegimen B - VerdiperstatRegimen C - TrehaloseRegimen D - PridopidineControl
InterventionZilucoplan 0.3 mg/kg SC dailyVerdiperstat 600 mg oral twice dailyTrehalose 0.75 g/kg IV weeklyPridopidine 45 mg oral twice dailyMatching placebo per regimen, pooled across concurrent regimens
Duration24 weeks24 weeks24 weeks24 weeks24 weeks

Outcomes

OutcomeTypeControlInterventionHR / OR / RRP-value
Disease Rate Ratio (DRR) from Bayesian shared-parameter joint model combining ALSFRS-R slope and survival; DRR <1 indicates slowing of disease progressionPrimaryShared placebo pool (42 regimen-specific + shared concurrent placebo per regimen)Pridopidine DRR 0.99 (95% CrI 0.80-1.21, probability <1 = 0.55); Verdiperstat DRR 0.98 (95% CrI 0.77-1.24, probability = 0.57); CNM-Au8 DRR 0.97 (95% CrI 0.783-1.175, probability <1 = 0.65); Zilucoplan DRR 1.08; Trehalose DRR 0.87None met prespecified efficacy threshold
CNM-Au8: Plasma neurofilament light chain (NfL) changeSecondaryIncreased 7.9% (placebo)Decreased 2.3% (CNM-Au8)p=0.04
Pridopidine: Completion rateSecondary84% completion rate over 24 weeks
Verdiperstat: Completion rateSecondary78% completion rate over 24 weeks
CNM-Au8: Completion rateSecondary90% completion rate over 24 weeks
Pridopidine - FallsAdverse29.3% (placebo)28.1% (pridopidine)
Pridopidine - Muscular weaknessAdverse31.7% (placebo)24.0% (pridopidine)
Verdiperstat - Key AEsAdverseNausea, insomnia, elevated thyrotropin
CNM-Au8 - DiarrheaAdverse7% (placebo)19% (CNM-Au8)
CNM-Au8 - NauseaAdverse8.6% (placebo)14.2% (CNM-Au8)

Subgroup Analysis

No prespecified subgroup analyses met efficacy thresholds. CNM-Au8 showed a significant biomarker effect on plasma NfL (p=0.04) despite no clinical DRR benefit.


Criticisms

  • None of the first five tested regimens (Zilucoplan, Verdiperstat, Trehalose, Pridopidine, CNM-Au8) demonstrated clinical efficacy
  • 24-week treatment duration may be insufficient for disease-modifying agents in ALS
  • Shared placebo design introduces statistical complexity and potential temporal confounding across regimens
  • US-only sites (54 centers) may limit generalizability to non-US ALS populations
  • Bayesian DRR endpoint not widely used outside this trial, limiting cross-study comparison
  • Platform master protocol allows concurrent enrollment which may create selection bias across regimens
  • CNM-Au8 biomarker signal (NfL reduction p=0.04) did not translate to clinical benefit, questioning NfL as a surrogate endpoint

Funding

Sean M. Healey and AMG Center for ALS, Tackle ALS, The ALS Association, Muscular Dystrophy Association

Based on: HEALEY ALS Platform Trial (Muscle & Nerve, 2025)

Authors: Harkey BA, Distefano S, Pagliaro JA, ..., Sherman AV

Citation: Muscle Nerve. 2025;72(2):294-305

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