Recruiting
Phase 3

Obudanersen

Sponsor:

Ionis Pharmaceuticals, Inc.

Code:

NCT07782827

Conditions

Angelman Syndrome

Eligibility Criteria

Sex: All

Age: 2 - 50

Healthy Volunteers: Not accepted

Interventions

obudanersen

Study Details

Brief summary:

The primary purpose of the study is to evaluate efficacy of obudanersen in participants with AS due to uniparental disomy or imprinting defects (UPD/ID) as measured through expressive communication.

Conditions

Angelman Syndrome

Study ID

NCT07782827

Start date

Sep, 2026

Status verified date

Sep, 2026

Completion date

May, 2031

Anticipated

Primary completion date

May, 2028

Anticipated

Eligibility Criteria

Eligibility Criteria

Sex: All

Age: 2 - 50

Healthy Volunteers: Not accepted

Key Inclusion Criteria:

1. Clinical diagnosis of AS with molecular result indicating either paternal UPD of 15q11.2-q13 or ID of the maternal 15q11.2-q13 region, provided by the Investigator and confirmed by either a qualified central vendor or a qualified local geneticist at the site.
2. The participant's caregiver(s)/legally-authorized representative (LAR) must have given written informed consent and any authorizations required by local law and be able and willing to comply with all study requirements.
3. Medically stable and can undergo sedation and/or general anesthesia without intubation.
4. Male or female between 2 and ≤ 50 years of age, depending on the specific cohort, at the time of the in-clinic Screening visit.
5. If applicable, is currently receiving stable doses of concomitant medications typically prescribed for AS, such as anti-epileptic medication, behavioral management medications, sleep medications, gabapentin, cannabidiol, and special diets, supplements, or nutritional support for at least 8 weeks prior to the Baseline visit. If recent changes (< 8 weeks stable) in medications, the participant may be allowed per Investigator judgment if the change is not expected to have an impact on the signs and symptoms of AS.
6. LAR/caregiver(s) agree(s) not to post any of the participant's personal medical data or information related to the study on any website or social media site (e.g., Facebook, Instagram, X, YouTube, TikTok, WhatsApp) from the time of enrollment until they are notified that the study is completed.

Key Exclusion Criteria:

1. Participant has a clinical diagnosis of AS with molecular confirmation of a UBE3A deletion or UBE3A mutation.
2. Any clinically significant abnormalities in medical history (e.g., major surgery within 3 months of Screening), or on physical examination for which treatment with an antisense oligonucleotide (ASO) would be contraindicated or which, in the opinion of the Investigator, could confound the results of this study.
3. Known brain or spinal disease that would interfere with the lumbar puncture (LP) procedure, cerebrospinal fluid (CSF) circulation, or presence of other factors that would affect the safety of the LP procedure, including tumors or abnormalities by MRI or computed tomography (CT), subarachnoid hemorrhage, suggestion of raised intracranial pressure (ICP) on magnetic resonance imaging (MRI) or ophthalmic examination, Chiari malformation, obstructive hydrocephalus, syringomyelia, tethered spinal cord syndrome, or connective tissue disorders such as Ehlers-Danlos syndrome and Marfan syndrome.
4. Any laboratory abnormalities or any other clinically significant abnormalities that would, as assessed by the Investigator, at Screening or Baseline, render a participant unsuitable for inclusion.
5. Previous treatment with an oligonucleotide (including small interfering ribonucleic acid \[siRNA\] and ASOs) or gene therapy or gene editing. This exclusion criterion does not apply to approved nucleic acid-based vaccines, including messenger Ribonucleic Acid (mRNA) vaccines, which are allowed.

Other inclusion/exclusion criteria may apply

Study Design

Enrollment

30 participants

Anticipated

Intervention Model

Single group

Primary purpose

Treatment

Interventions and Outcome Measures

Arms

experimental: obudanersen 80 mg

Participants in Cohorts 1 and 2 will be administered obudanersen via intrathecal (IT) bolus injection every 12 weeks during the Treatment Evaluation and LTE periods.

Interventions

obudanersen

ION582 will be administered by IT injection.

Primary outcome measure

  • Change From Baseline in Performance on the Expressive Communication Domain Raw Score Without Caregiver Input of the Bayley Scales for Infant and Toddler Development-4 (Bayley-4) in Cohort 1 [ Time Frame: Baseline and Week 52 ]

Central Contacts and Locations

Central contacts

Locations

University of North Carolina at Chapel Hill School of Medicine

Recruiting

Carrboro, North Carolina, United States, 27510

More Information

Sponsor

Ionis Pharmaceuticals, Inc.

Last update posted

Sep 28, 2026

Last verified

Sep, 2026

Keywords

  • Paternal Uniparental Disomy
  • ION582
  • imprinting defects

Trial information was received from ClinicalTrials.gov and was last updated on 2026-09-29. This information was provided to ClinicalTrials.gov by Ionis Pharmaceuticals, Inc. on 2026-09-28. Recruitment status is synced daily from ClinicalTrials.gov and may not reflect the sponsor's current status. Confirm during your call.