RecruitingPhase 1Phase 2NCT07215416

Safety and Efficacy of Mutation-targeted Precision Genetic Therapy for Ataxia-Telangiectasia (A-T)

A Phase 1/2 Study of Antisense Oligonucleotide Therapy for Treatment of Ataxia-Telangiectasia


Sponsor

Timothy Yu

Enrollment

10 participants

Start Date

Aug 1, 2026

Study Type

INTERVENTIONAL

Conditions

Summary

This project aims to evaluate the safety and efficacy of precision genetic therapy for patients with Ataxia-telangiectasia (A-T), a rare neurodegenerative disease caused by mutations in the ATM gene. The investigators will conduct a clinical trial to study the safety and efficacy of intrathecal administration of atipeksen, a targeted genetic therapy that restores ATM gene function in A-T individuals bearing the recurrent ATM c.7865C\>T variant. The aim of this study is to delay or forestall progression of neurologic symptoms in A-T and improving quality of life. Success will provide an empirical foundation for advancing additional precision genetic therapies for A-T and other neurodegenerative conditions.


Eligibility

Min Age: 0 YearsMax Age: 17 Years

Exclusion Criteria15

  • Who can take part:
  • People with classic A-T confirmed by genetic testing
  • Must have a specific ATM gene change (c.7865C>T)
  • Must also have another ATM change that causes A-T
  • Who cannot take part:
  • People with health problems that make lumbar puncture unsafe:
  • Blood clotting or bleeding problems
  • Brain conditions raising pressure inside the head
  • Serious heart or breathing problems
  • Infection near the lower back
  • Other things doctors will check:
  • Overall health and stability
  • Any medicines that might cause problems
  • Past difficulties with lumbar punctures
  • Any other safety concerns

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Interventions

DRUGAntisense oligonucleotide targeting the ATM gene

Atipeksen is a fully modified PS-2'MOE splice-switching antisense oligonucleotide that is designed to restore normal splicing patterns in patients with the ATM c.7865C\>T mutation.


Locations(1)

Boston Children's Hospital

Boston, Massachusetts, United States

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NCT07215416


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