Precision Genetic Therapy for Ataxia-Telangiectasia (A-T)

This study is testing a new treatment called atipeksen for children and teenagers with Ataxia-Telangiectasia (A-T), a rare brain disease. Atipeksen is a genetic therapy designed to fix a specific problem in the ATM gene (c.7865C>T mutation) that causes A-T. Researchers want to see if atipeksen is safe and if it can slow down or stop the worsening of brain symptoms in people with A-T. Success would mean improved quality of life. You can join if you are 0-17 years old, have A-T confirmed by genetic testing, and have the specific ATM gene change (c.7865C>T) along with another ATM change. You cannot join if you have certain health problems that make a spinal tap (lumbar puncture) unsafe. The study plans to enroll 10 participants.

Study design
This is an interventional study, but the phase is not specified. It aims to enroll 10 participants.
What's involved
Neurological function will be measured at the start and every 12 weeks for up to ten years.
Compensation
Not stated in the trial record.
Follow-up
Participants will be followed for neurological function and global impression of change for up to ten years.

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NCT07215416

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

Recruiting
PHASE1Ages 0–17InterventionalTreatment
Timothy Yu
~10 participants
Updated 2026-05-28 on ClinicalTrials.gov
What's tested:Antisense oligonucleotide targeting the ATM gene

At a glance

Recruiting sites
1 of 1 listed site is recruiting right now
RecruitingSuspended, closed, or not yet open
What they're measuring
Neurological function as measured by the AT-NEST scale
Measured over At Baseline and every 12 weeks up to ten years
+1 more outcome measured
Ataxia Telangiectasia
1 sites across 1 states
Massachusetts1
  • Timothy Yu, MD, PhD · STUDY_DIRECTOR · Boston Childrens Hostpital
  • Christelle Achkar, MD · PRINCIPAL_INVESTIGATOR · Boston Children's Hospital

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Eligibility criteria

Inclusion

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
Blood clotting or bleeding problems
Brain conditions raising pressure inside the head
Serious heart or breathing problems
Infection near the lower back
Overall health and stability
Any medicines that might cause problems
Past difficulties with lumbar punctures
Any other safety concerns
  • Neurological function as measured by the AT-NEST scaleAt Baseline and every 12 weeks up to ten years

    The Ataxia-Telangiectasia Neurological Examination Toolkit (AT-NEST) is a test designed specifically for people with Ataxia-Telangiectasia (A-T). It helps doctors and researchers understand how the brain and nerves are working. The test looks at six main areas: * Communication * Eye movements Ataxia (problems with balance and coordination) * Movement disorders * Muscle strength (Power) * Nerve function (Neuropathy) These six areas make up the "pure neuro" score, which ranges from 0 (lowest) to 100 (highest). The test also includes growth and nutrition. When these are added to the six main areas, a total "neuro-related" score is calculated, with a maximum of 114. Higher scores mean better overall performance.This test is not painful and is carried out by trained specialists who will guide you through each step.

  • Ataxia-Telangiectasia Structured Clinical Global Impression of Change (A-T CGI)At Baseline and every 12 weeks up to ten years

    The Ataxia-Telangiectasia Clinical Global Impression of Change (AT-CGI) is a tool used to track how A-T progresses over time. It looks at five main areas of the disease: Ataxia (balance and coordination problems) Dysarthria (difficulty speaking) Repetitive movements / Dysmetria (trouble controlling movements) Movement disorders Eye movements Each area is scored from 0 to 4, for a total of 20 points. Higher scores mean more severe symptoms, while lower scores indicate better function.