Safety and Efficacy of DYNE-251 in Males with DMD Mutations Amenable to Exon 51 Skipping
Perry Shieh1, Craig Campbell2, Nicolas Deconink3, Liesbeth De Waele4, Kevin Flanigan5, Michelle Lorentzos6, Han Phan7, Chris Mix8, Soma Ray8, Dazhe Wang8, Ashish Dugar8, Douglas Kerr8, Maria Naylor8, Michela Guglieri9
1University of California Los Angeles, 2London Health Sciences Centre, 3Neuromuscular Reference Center UZ Gent, 4University Hospitals Leuven, 5Nationwide CHildrens Hospital, 6Children's Hospital at Westmead, 7Rare Disease Research, LLC, 8Dyne Therapeutics, Inc., 9Royal Victoria Infirmary, Newcastle University
Objective:
Determine the safety and efficacy of DYNE-251 in the Phase 1/2 DELIVER trial (NCT05524883) of males aged 4-16 with DMD mutations amenable to exon 51 skipping.
Background:

DMD is caused by absence of functional dystrophin protein. DYNE-251, an investigational therapeutic for DMD, leverages the transferrin receptor to deliver an exon 51-skipping PMO to affected tissues with the goal of restoring dystrophin expression.

Design/Methods:

In the MAD portion of DELIVER, participants are randomized to receive DYNE-251 or placebo Q4W or Q8W for 6 months and are subsequently escalated to the highest tolerable dose in the OLE/LTE. All 54 currently enrolled participants are at the 20 mg/kg dose level. 

Results:

DYNE-251 drove dose-dependent increases in mean PMO muscle concentration and exon skipping, resulting in 3.22% and 3.72% of normal mean dystrophin in the 10 mg/kg and 20 mg/kg cohorts, respectively, at 6 months. The mean corresponding muscle content-adjusted dystrophin levels were 7.64% and 8.72% of normal. DYNE-251 led to improvements across multiple functional endpoints, including the North Star Ambulatory Assessment, Time to Rise from Floor, 10-Meter Walk/Run Test, and Stride Velocity 95th Centile. Improvements were noted as early as 6 months in both the 10 mg/kg and 20 mg/kg cohorts; a continued effect through 12 months was observed in the 10 mg/kg cohort. DYNE-251 had a favorable safety profile as of the analysis date with most TEAEs reported as mild or moderate. Three serious TEAEs potentially related to study drug occurred in two participants in the 40 mg/kg Q4W cohort. These events had multiple confounding factors suggestive of infection or background risk that may have contributed to their presentation. Subsequently, all participants in the 40 mg/kg cohorts were lowered to the 20 mg/kg dose level.

Conclusions:

DYNE-251 had a favorable safety profile and resulted in early improvements across multiple functional endpoints.

10.1212/WNL.0000000000211198
Disclaimer: Abstracts were not reviewed by Neurology® and do not reflect the views of Neurology® editors or staff.