Neurology at Forefront of FDA’s Complex Innovative Trial Design Pilot

The coronavirus pandemic has frozen many clinical trials and forced others to adapt in the face of overwhelmed healthcare systems around the world. When activity resumes, trials could look different, with more remote monitoring and decentralized enrollment.

They could reflect some of the design evolutions that the US Food and Drug Administration and sponsors have been exploring as part of the complex innovative trial design pilot—and will be put to the test during the development COVID-19 vaccines and therapeutics.

The first two proposals accepted into the complex innovative trial design program (CID) by the FDA target central nervous system conditions: Duchenne muscular dystrophy and chronic pain.

The program is committed to engaging sponsors across therapeutic areas, but it is perhaps not surprising that the early adopters come from neurology. CNS drug development spent much of the last decade in the doldrums, but the era marked by the introduction of the breakthrough therapy designation has seen neurology rise to become one of the most active and most innovative areas after oncology.

Innovative drugs sometimes require innovative trial designs, which are often complicated by the need to rely on patient-reported outcomes and clinical outcome assessments (COAs) as endpoints in clinical trials. COAs have been a major focus of the FDA’s efforts to improve regulatory science.

The CID pilot program, a joint effort of the Center for Drug and Center for Biologics Evaluation and Research, was mandated by the 21st Century Cures Act/PDUFA VI. The CID pilot meeting program kicked off on 30 August 2018 and will run through 30 September 2022. Participants will have the opportunity for two focused meetings with FDA staff.

While the limited number of CID pilot announcements suggests that uptake into the program has started out slowly, sponsors are not required to disclose their participations—although sponsors must agree to FDA’s public disclosure of study design and analysis elements.

The disclosure requirement had been a sticking point for adoption of the pilot, but the agency issued a Federal Register notice in August 2019 clarifying that disclosure would not include the sponsor or product name, subject-level data, recruitment strategies, or comprehensive eligibility criteria.

The FDA has ramped up promotion of the CID initiative over 2019, including the mid-summer release of a CID presentation series. The agency will select up to two requests per quarter over the five-year course of the pilot.

“FDA will give priority consideration for CID Pilot Meeting Program inclusion to proposals with highly innovative trial designs for which analytically derived properties might not be feasible and for which simulations are necessary to determine design operating characteristics,” the CID presentation reports, pointing to the potential to address the risk of type I error, or “the probability of falsely concluding that a medical product has an effect when it does not.”

The pilot meeting program will also evaluate the level of medical need that the CID would address. CIDs could be useful in rare diseases, FDA suggests, and for programs studying “multiple body sites in anti-infective drug development.”

The agency released a draft guidance on interacting with FDA on complex innovative trial designs in September 2019. The agency CID meeting pilot has an ambitious timeline for sponsor interaction, aiming to schedule both of the pilot’s sponsor meetings within eight months.

FIRST WAVE

Wave Life Sciences (NASDAQ: WVE) became the first sponsor to go public with a CID pilot acceptance in January 2019, for a Phase 2/3trial of the exon-skipping candidate suvodirsen in Duchenne muscular dystrophy (DMD) patients amenable to exon 51 skipping. By the end of the year, though, the firm stopped development after interim analysis of a Phase 1 open-label extension study showed no change from baseline in dystrophin expression.

Eli Lilly (NYSE: LLY) joined the CID pilot with a proposed “master protocol for the development of novel approaches to the treatment of multiple types of chronic pain,” the company announced 5 September 2019.

Lilly’s trial “will evaluate multiple interventions in several chronic pain conditions via one streamlined clinical protocol,” the company reported. “The learning from this CID will contribute to broader utilization of the innovative statistical approaches required.”

Lilly is one of the major players in the pain market, offering migraine therapies both chronic (the CGRP [antag] Emgality) and acute (Reyvow). A potential blockbuster pain candidate, tanezumab, is well into a broad Phase 3 program targeting osteoarthritis pain, cancer pain, and chronic low back pain.

Lilly has disclosed three novel candidates for treatment of pain in Phase 1, including a potential first-in-class non-opioid agent for chronic pain from a collaboration with AC Immune SA. The somatostatin receptor type 4 agonist was selected as a “Phase 1 highlight” in Lilly’s third quarter 2019 earnings call. An internal readout is expected in 2020 to “inform potential progression to Phase 2,” the company said.

Phase 1 studies of the PACAP38 monoclonal antibody LY3451838 and the TRPA1 antagonist LY3526318 are expected to complete in early 2020.

The innovative features of Wave’s Phase II/III DYSTANCE 51 trial focus on reducing the number of patients that must be enrolled in order to deliver conclusive evidence of clinical efficacy, the company explained in its 5 November 2019 quarterly filing with the Securities and Exchange Commission.

Under the pilot, the company aims to minimize “the number of patients required in the placebo treatment arm” and, hopefully, accelerate completion of the trial.

DYSTANCE 51 began enrolling boys between 5 and 12 years old with genetically confirmed DMD amenable to exon 51 skipping therapy in June 2019. The primary efficacy endpoints will assess change in dystrophin protein level and change in the North Star Ambulatory Assessment score. “Multiple functional outcome measures” serve as secondary endpoints.

“We intend to use the results of this trial to seek regulatory approvals globally,” Wave stated.

An open-label extension of a Phase 1 trial has been collecting data from quarterly clinical outcome measures while taking muscle biopsies. An interim analysis will