Gene Therapy Trial for Otoferlin Gene-mediated Hearing Loss

NCT ID: NCT05821959

Last Updated: 2026-01-15

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

RECRUITING

Clinical Phase

PHASE1/PHASE2

Total Enrollment

22 participants

Study Classification

INTERVENTIONAL

Study Start Date

2023-09-15

Study Completion Date

2028-10-31

Brief Summary

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Part A of this trial will evaluate the safety and tolerability of a single surgical administration procedure in one or both ear(s) with one of two dose levels of AAVAnc80-hOTOF and will evaluate the Akouos Delivery Device, together with the Precision Delivery Mechanism, to safely achieve the intended product performance.

Detailed Description

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The following two dose levels will be tested:

* Up to 4.1E11 total vg/cochlea
* Up to 8.1E11 total vg/cochlea

Conditions

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Sensorineural Hearing Loss, Bilateral

Study Design

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Allocation Method

NON_RANDOMIZED

Intervention Model

SEQUENTIAL

Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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Cohort 1a and Cohort 1b

Cohort 1a: Two participants aged 7 to 17 years old (inclusive) to receive intracochlear administration of AAVAnc80-hOTOF (dose level 1) in the study ear using a sterile, one-time use investigational medical device

Cohort 1b: Up to six eligible participants will be enrolled to receive intracochlear administration of AAVAnc80-hOTOF (dose level 1) in the study ear using a sterile, one-time use investigational medical device; the first three participants will be at least two years of age at the time of AAVAnc80-hOTOF administration and the next three participants may be of any age

Group Type EXPERIMENTAL

AAVAnc80-hOTOF via Akouos Delivery Device

Intervention Type COMBINATION_PRODUCT

AAVAnc80-hOTOF is a sterile suspension intended to be administered via a one-time unilateral intracochlear administration using the Akouos Delivery Device.

Cohort 2

Cohort 2: Up to six eligible participants will be enrolled to receive intracochlear administration of AAVAnc80-hOTOF (dose level 2) in the study ear using a sterile, one-time use investigational medical device; the first three participants will be at least two years of age at the time of AAVAnc80-hOTOF administration and the next three participants may be of any age

Group Type EXPERIMENTAL

AAVAnc80-hOTOF via Akouos Delivery Device

Intervention Type COMBINATION_PRODUCT

AAVAnc80-hOTOF is a sterile suspension intended to be administered via a one-time unilateral intracochlear administration using the Akouos Delivery Device.

Bilateral Expansion

Bilateral Expansion: up to eight eligible participants of any age, and no cochlear implants, may be eligible for enrollment into the Bilateral Expansion Cohort (simultaneous bilateral administration of AAVAnc80-hOTOF).

Group Type EXPERIMENTAL

AAVAnc80-hOTOF via Akouos Delivery Device and Precision Delivery Mechanism

Intervention Type COMBINATION_PRODUCT

AAVAnc80-hOTOF is a sterile suspension intended to be administered via a one-time bilateral intracochlear administration using the Akouos Delivery Device together with the Precision Delivery Mechanism.

Interventions

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AAVAnc80-hOTOF via Akouos Delivery Device

AAVAnc80-hOTOF is a sterile suspension intended to be administered via a one-time unilateral intracochlear administration using the Akouos Delivery Device.

Intervention Type COMBINATION_PRODUCT

AAVAnc80-hOTOF via Akouos Delivery Device and Precision Delivery Mechanism

AAVAnc80-hOTOF is a sterile suspension intended to be administered via a one-time bilateral intracochlear administration using the Akouos Delivery Device together with the Precision Delivery Mechanism.

Intervention Type COMBINATION_PRODUCT

Eligibility Criteria

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Inclusion Criteria

1. Participants may be of any age, based on Cohort Criteria
2. At least two mutations in the otoferlin gene
3. Clinical presentation of Profound bilateral sensorineural hearing loss as assessed by ABR
4. Preserved distortion product otoacoustic emissions (DPOAEs)
5. Able and willing to comply with all study requirements, including willingness to participate in a separate long term follow-up study after completion of this trial

Criteria for Exclusion:

1. Persistent ear infections, anatomic or other abnormalities of the ear, and/or medical conditions that would contraindicate undergoing surgery, anesthesia, and/or administration of investigational gene therapy
2. Cochlear Implant(s) in the ear(s) to receive AAVAnc80-hOTOF
3. Prior participation in a clinical trial with an investigational drug, within six months prior to administration, or any prior participation in a gene therapy clinical trial
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Eli Lilly and Company

INDUSTRY

Sponsor Role collaborator

Akouos, Inc.

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Kathy Reape, MD

Role: STUDY_DIRECTOR

Akouos, Inc.

Locations

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University of California, San Francisco Benioff Children's Hospital

San Francisco, California, United States

Site Status RECRUITING

University of Iowa

Iowa City, Iowa, United States

Site Status RECRUITING

Boston Children's Hospital

Boston, Massachusetts, United States

Site Status RECRUITING

Cincinnati Children's Hospital Medical Center

Cincinnati, Ohio, United States

Site Status RECRUITING

Children's Hospital of Philadelphia

Philadelphia, Pennsylvania, United States

Site Status RECRUITING

Vanderbilt Bill Wilkerson Center

Nashville, Tennessee, United States

Site Status RECRUITING

The Hospital for Sick Children [email protected] +1 (416) 813 6683

Toronto, Ontario, Canada

Site Status RECRUITING

National Taiwan University Hospital

Taipei, , Taiwan

Site Status RECRUITING

University College London

London, United Kingdom, United Kingdom

Site Status RECRUITING

Countries

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United States Canada Taiwan United Kingdom

Central Contacts

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Akouos Clinical Trials

Role: CONTACT

+1 857-410-1816

Facility Contacts

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Jenny Stephans

Role: primary

415-353-2012

Kesten Anderson

Role: primary

+1 319-356-8320

Meghan Fitzgerald

Role: primary

+1 617-355-4729

Alexandra Sestito

Role: primary

+1 513 636-4355

Aminat Adegbuji

Role: primary

+1 267-426-8380

Taha Jan, MD

Role: primary

+1 615-421-1228

Jaina Negandhi

Role: primary

+1 416-813-6683

Wan-Cian Liao

Role: primary

+886- 953-728-657

Role: primary

Other Identifiers

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AK-OTOF-101

Identifier Type: -

Identifier Source: org_study_id

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