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Active, not recruitingNCT06617429AspireUpdated Aug 6, 2026

Phase 3 Efficacy and Safety Study of GTX-102 in Pediatric Subjects With Angelman Syndrome (AS)

A Phase 3 interventional study of GTX-102 and Sham-LP in Angelman Syndrome, sponsored by Ultragenyx Pharmaceutical Inc. Active, not recruiting at 28 sites in 6 countries. Open to participants aged 4 Years to 17 Years. Per ClinicalTrials.gov, last updated 2026-08-06.

Sponsored by Ultragenyx Pharmaceutical Inc · Phase 3, Interventional, and Treatment

Phase
Phase 3
Study type
Interventional
Enrollment
129
Allocation
Randomized
Ages
4 Years to 17 Years
Sex
All
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Study summary

The primary objective of this study is to evaluate the effect of GTX-102 in cognitive function in participants with deletion-type Angelman Syndrome (AS).

02

Conditions studied

  • Angelman Syndrome

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Keywords

  • Angelman Syndrome
03

In context

Angelman Syndrome

46 studies on the registry are indexed under Angelman Syndrome; 16 are open to participants now.

This study's enrollment of 129 is above the median of 67 across 25 interventional studies indexed under Angelman Syndrome.

Browse Angelman Syndrome studies →

Lead sponsor

Ultragenyx Pharmaceutical Inc is the lead sponsor of 63 studies on the registry; 8 are open to participants now.

Of its 13 completed or terminated interventional studies of FDA-regulated products, 11 (85%) have results posted.

Counted across the registry records on this site, refreshed daily.

04

Who can participate

Ages eligible
4 Years to 17 Years
Sexes eligible
All
Accepts healthy volunteers
No

Eligibility criteria

Key Inclusion Criteria:

  • Signed informed consent from parent(s) or legal guardian(s)
  • Confirmed diagnosis of AS with genetic confirmation of full maternal ubiquitin-protein ligase E3A (UBE3A) gene deletion causing AS in the region of 15q11.2 q13
  • Able to ambulate independently, or with assistance at the Screening Visit (note, a child whose primary means of mobility is by wheelchair is excluded from the study)
  • Platelet count, prothrombin time / international normalized ratio, and partial thromboplastin time within 1.5x the normal limits at the Screening Visit
  • Willing and able to comply with scheduled visits, drug administration plan, laboratory tests, and all study procedures, including LP procedure, MRI, and tolerating anesthesia without intubation
  • From the time of informed consent through to at least 6 months after the final dose of GTX-102, females of childbearing potential who are sexually active must use highly effective contraception or abstinence. Males are able to participate if they agree to remain abstinent (refrain from heterosexual intercourse) or use acceptable contraceptive methods during the study and for at least 3 months after the final dose of GTX-102

Key Exclusion Criteria:

  • Any change in medications or diet/supplements intended to treat symptoms of AS (eg, sleeping aids, antiseizure medications, supplements, dietary change including ketogenic or low-glycemic index diet, other) within the month prior to the Screening Visit (excluding weight-based adjustments)
  • Any condition that creates an increased risk of unsuccessful LP
  • Current or expected concomitant use of drugs that increase the risk of bleeding (eg, heparin, low molecular weight heparin, platelet inhibitors)
  • Known hypersensitivity to GTX-102 or its excipients that, in the judgment of the Investigator, places the subject at increased risk for adverse effects
  • Presence or history of any condition, lab abnormality, or infection, that, in the judgement of the Investigator, would interfere with participation, pose undue safety risk, or would confound interpretation of results
  • Pregnant or breastfeeding or planning to become pregnant (self or partner) at any time during the study
  • Use of any investigational product or investigational medical device within 6 months or 5 half-lives prior to the Screening Visit or any prior use of gene therapy or ASO regardless of duration since last administration
  • Concurrent participation in any interventional study
05

Study design

Phase
Phase 3
Primary purpose
Treatment
Allocation
Randomized
Intervention model
Parallel assignment
Masking
Quadruple (Participant, Care provider, Investigator, Outcomes assessor)
Enrollment
129 participants (actual)

Study arms

  • Experimental
    GTX-102

    Participants will receive GTX-102 via lumbar puncture (LP) during both the double-blind and open-label period

    Drug: GTX-102

  • Sham comparator
    Sham-LP then GTX-102

    Participants will receive sham procedure during the double-blind period and then will receive GTX-102 via LP during the open-label period

    Drug: GTX-102 · Procedure: Sham-LP

Interventions

  • DrugGTX-102

    antisense oligonucleotide

    Also known as: apazunersen

  • ProcedureSham-LP

    Small needle prick on the lower back at the location where the LP injection is normally made

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What researchers measure

Primary outcomes

  1. Change from Baseline in Bayley-4 Cognitive Raw Score Without Caregiver Input at Day 338

    Time frame: Baseline, Day 338

Secondary outcomes

  1. Net Response in Multidomain Responder Index (MDRI)

    Time frame: Day 338

  2. Change from Baseline in ABC-C Hyperactivity/Noncompliance Subscale Score at Day 338

    Time frame: Baseline, Day 338

  3. Change from Baseline in Bayley-4 Receptive Communication Raw Score at Day 338

    Time frame: Baseline, Day 338

  4. Change from Baseline in Angelman Severity Assessment (ASA) Sleep Rating Raw Score at Day 338

    Time frame: Baseline, Day 338

  5. Change from Baseline in Vineland Adaptive Behavior Scales-3 (Vineland-3) Receptive Communication Raw Score at Day 338

    Time frame: Baseline, Day 338

  6. Change from Baseline in Vineland-3 Expressive Communication Raw Score at Day 338

    Time frame: Baseline, Day 338

  7. Change from Baseline in Bayley-4 Gross Motor Raw Score at Day 338

    Time frame: Baseline, Day 338

  8. Change from Baseline in ASA Gross Motor Rating at Day 338

    Time frame: Baseline, Day 338

  9. Number of Participants with Treatment Emergent Adverse Events (AEs) and Serious Adverse Events (SAEs), Severity of AEs and Relationship to Investigational Drug, Procedure, and Premedication

    Time frame: 2 Years

07

Study locations

28 sites
  • Cedars Sinai
    Los Angeles, California 90048, United States
  • UCSD, Rady Children's Hospital
    San Diego, California 92123, United States
  • UCSF
    San Francisco, California 94143, United States
  • Children's Hospital Colorado
    Aurora, Colorado 80045, United States
  • Nicklaus Children's Hospital
    Miami, Florida 33155, United States
  • Rare Disease Research
    Atlanta, Georgia 30329, United States
  • Rush University
    Chicago, Illinois 60612, United States
  • Boston Children's Hospital
    Boston, Massachusetts 02115, United States
  • Children's Mercy
    Kansas City, Missouri 64108, United States
  • Columbia University Medical Center
    New York, New York 10032, United States
  • UNC Chapel Hill Pediatrics
    Chapel Hill, North Carolina 27599, United States
  • Rare Disease Research
    Hillsborough, North Carolina 27278, United States
  • The University of Texas
    Austin, Texas 78723, United States
  • Carum Research Inc
    Dallas, Texas 75243, United States
  • McGill University Health Centre
    Montreal, Quebec H4A3J1, Canada
  • British Columbia Children's Hospital
    Vancouver, V6H 3V4, Canada
  • Universitaetsklinikum Hamburg-Eppendorf
    Hamburg, 20251, Germany
  • University of Leipzig
    Leipzig, 04155, Germany
  • Haunersche Kinderklinik
    Munich, 80336, Germany
  • Nagoya City University Graduate School of Medical Sciences
    Nagoya, Aichi-ken 467-0001, Japan
  • Osaka City General Hospital
    Osaka, 543-0021, Japan
  • Hokkaido University Hospital
    Sapporo, 060-8648, Japan
  • Medical University of Gdańsk
    Gdansk, 80211, Poland
  • Polish Mothers Memorial Institute
    Lodz, 93-338, Poland
  • Hospital Universitario Parc Tauli
    Barcelona, 08208, Spain
  • Hospital Sant Joan de Deu
    Barcelona, 08950, Spain
  • Hospital Universitario Puerta de Hierro
    Madrid, 28222, Spain
  • Hospital Universitario Virgen del Rocio
    Seville, 41013, Spain
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References and documents

09

Updates

Tracking since Sep 25, 2026
No changes since tracking began. The registry record was last updated on Aug 6, 2026, before this site started recording changes on Sep 25, 2026. Its history is on ClinicalTrials.gov ↗
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Registry details

Key details

Study ID
NCT06617429
Lead sponsor
Ultragenyx Pharmaceutical Inc
Responsible party
Sponsor
First posted
Sep 27, 2024
Start date
Dec 3, 2024
Primary completion
Jul 10, 2026
Completion
Nov 2027 (estimated)
Last update
Aug 6, 2026

Study contacts

Medical Director
study director · Ultragenyx Pharmaceuticals Inc.

Oversight

FDA-regulated drug
Yes
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

Not currently enrolling

This study is active, not recruiting, as verified in Aug 2026. You cannot join it, but the record below documents what was studied.

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