A Rollercoaster Ride to Hope
The AMT-130 Story, and What the FDA's June 2026 Announcement Means for Our Community
Our community’s first meeting with the pharmaceutical company uniQure took place in September 2018. At the time, the idea that a gene therapy could actually slow HD felt like pie in the sky. Nearly eight years later, on June 17, 2026, that hope took a major step toward becoming real.
This update walks through that journey — the excitement, the setbacks, and the advocacy that helped get us here — and what it means going forward.
What is AMT-130?
Gene therapy aims to treat disease by changing the genetic instructions inside our cells. Imagine your DNA is a cookbook. If one recipe contains a mistake, gene therapy works like an editor, correcting or blocking the faulty recipe.
AMT-130 is designed to reduce production of the harmful huntingtin protein that causes HD. It uses a harmless virus to deliver a small genetic molecule into the brain that “switches down” the faulty huntingtin gene. The treatment is given once through MRI-guided brain surgery and has been tested at both a low and a high dose.
So far, AMT-130 has been studied in two Phase I/II clinical trials:
- United States: Began in June 2020. Participants were randomly assigned to receive either AMT-130 or a sham (imitation) brain surgery.
- Europe: Began in February 2022 in Warsaw, Poland, and later expanded to sites in the UK. Everyone in this study received AMT-130.
The Journey So Far
- September 24, 2025 – Landmark results
Three years after treatment, people who received the high dose of AMT-130 showed a 75% slowing in disease progression compared with matched external controls alongside favourable biomarker changes. You can read more here. - November 3, 2025 – A difficult reversal
Following an October 29 pre-submission meeting, the FDA told uniQure it no longer agreed that the trial data — compared against that matched “external control” group rather than an internal placebo group — were sufficient, on their own, to support a Biologics License Application (BLA), the formal request to market AMT-130 in the US.
This was a major disappointment. A new trial would require more volunteers to undergo brain surgery without receiving the treatment, raising serious ethical concerns for this fatal, progressive disease where every year of delay matters. - Late 2025 to January 2026 – The community responds
As they say, “you can’t keep a good man down”: patient advocacy groups in the US quickly mobilised and started 2 petitions which were signed worldwide. On January 22, 2026, with the petitions having gathered more than 48,000 signatures, representatives hand-delivered them to FDA alongside more than 11,000 messages sent to Congress.The petitions called on the FDA to honour its prior guidance and expedite review by allowing uniQure to file a BLA for accelerated approval consideration for its investigational gene therapy, AMT-130
Community representatives also secured a place in discussions with the FDA, helping ensure patients’ voices were heard. - March 2, 2026 – The setback confirmed
The FDA confirmed its position, saying the existing data were still not enough for approval and again recommending a new randomized, sham-controlled trial. - April 30, 2026 – Progress in the UK
While discussions continued in the US, uniQure announced a positive meeting with the UK’s Medicines and Healthcare products Regulatory Agency (MHRA). The company confirmed plans to apply for UK approval in the third quarter of 2026. - June 17, 2026 – A turning point
In a major reversal, the FDA and uniQure reached a breakthrough alignment. The FDA agreed to allow uniQure to submit an application for accelerated approval based on the totality of their existing Phase I/II data and three-year follow-up, bypassing the need for a highly controversial sham-surgery study. Read the press release here.
What Happens Next?
It’s important to understand what this does—and doesn’t—mean.
- AMT-130 is still experimental. It has not yet been approved and is only available through clinical trials.
- The FDA application has not yet been submitted. uniQure expects to file it in Q3 2026, after which the FDA review process will begin.
- Accelerated approval is conditional. If granted, it would allow earlier patient access while requiring a follow-up study to confirm the treatment’s long-term benefit.
- The UK review is happening separately. uniQure also plans to submit its application to the MHRA during Q3 2026.
Even with these important steps still ahead, June 17, 2026 represents a genuine milestone. The achievements are thanks to the trial participants who volunteered for brain surgery, the researchers who continued pushing forward, and a community that refused to give up when the path became more difficult.
For the first time, a treatment has shown in people that it may be possible to slow the progression of Huntington’s disease.
– Article written by Dina de Sousa
