◎ Investigational

AMT-130 (AAV5-miHTT gene therapy)

1
Evidence entries
2
Clinical trials
Overview
Treatment Details
Mechanism of Action
A one-time gene therapy delivered via a surgical procedure directly into the brain, using an AAV5 viral vector to carry a microRNA construct that silences the huntingtin gene, reducing mutant huntingtin production at the source. Positive 36-month Phase 1/2 follow-up data showed slowed disease progression and reduced cerebrospinal fluid neurofilament light (NfL), a biomarker of nerve damage. uniQure met with the FDA to discuss a Biologics License Application (BLA) filing.
Dosing Notes
Investigational; one-time neurosurgical intraparenchymal infusion (not repeat dosing).
Treatment Type
pharmaceutical
Clinical Evidence
What the Evidence Shows
Each finding below is linked to its primary source. Confidence levels reflect the quality and quantity of available evidence — not CelluTarget's endorsement of any treatment.
High: Supported by multiple robust studies or regulatory approval
Moderate: Supported by limited controlled studies or consistent case series
Low: Based on case reports, expert opinion, or early-phase data only
Moderate ConfidenceSupported by limited controlled studies or consistent case series
Positive 36-month follow-up data from the Phase 1/2 trial showed that roughly 80% of patients who received AMT-130 had slowed disease progression, along with reductions in cerebrospinal fluid neurofilament light (NfL), a biomarker of nerve damage. Results are based on fewer than 30 participants with comparisons to external controls, so some caution is warranted given the small, non-randomized-controlled nature of the readout. uniQure met with the FDA in 2025 and aims to file for approval in early 2026.
Verified Jul 2026
Institutional Research
The First Domino Falls: AMT-130 Gene Therapy Slows Huntington's in Landmark Trial — HDBuzz
Published January 2026
View source ↗
Clinical Trials
Linked Trials2 trials
AMT-130 (AAV5-miHTT gene therapy) for MMN — Evidence & Clinical Trials | CelluTarget