A Study of Adeno-Associated Virus Serotype 8-Mediated Gene Transfer of Glucose-6-Phosphatase in Patients With Glycogen Storage Disease Type Ia (GSDIa)

NCT ID: NCT05139316

Last Updated: 2025-11-19

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

ACTIVE_NOT_RECRUITING

Clinical Phase

PHASE3

Total Enrollment

49 participants

Study Classification

INTERVENTIONAL

Study Start Date

2021-11-08

Study Completion Date

2026-02-28

Brief Summary

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The primary objectives of this study are to evaluate the efficacy of DTX401 to reduce or eliminate dependence on exogenous glucose replacement therapy to maintain euglycemia and to maintain or improve the quality of glucose control.

Detailed Description

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Study DTX401-CL301 is a phase 3 study to determine the efficacy and confirm the safety of DTX401 in patients 8 years and older with glycogen storage disease type Ia (GSDIa).

Participants will be randomized 1:1 to DTX401 or placebo group, and followed closely for 48 weeks. At week 48 eligible participants will cross over and receive DTX401 if they had previously received placebo or placebo if they had previously received DTX401, and will be followed closely for an additional 96 weeks. After completion of week 144 or early withdrawal, participants will be offered enrollment into a Disease Monitoring Program (DMP) where they will be followed for at least 10 years post DTX401 infusion.

In Japan, there will be a single open label study arm and all participants will be treated with DTX401. At week 48, Japanese participants will be offered enrollment into a Disease Monitoring Program (DMP) where they will be followed for at least 10 years post DTX401 infusion.

Conditions

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Glycogen Storage Disease Type IA

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

CROSSOVER

Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors

Study Groups

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DTX401, Then Placebo

Participants receive single peripheral intravenous (IV) infusion of DTX401 in solution. At week 48 participants receive single peripheral IV infusion of Placebo.

Group Type EXPERIMENTAL

DTX401

Intervention Type GENETIC

nonreplicating, recombinant, adeno-associated virus (AAV) serotype 8 (AAV8)

Placebo

Intervention Type OTHER

Normal Saline infusion

Oral prednisolone

Intervention Type DRUG

Participants who receive DTX401 solution will receive oral prednisolone

Placebo for oral prednisolone

Intervention Type DRUG

Participants who receive placebo will receive placebo oral prednisolone to maintain the study blind

Placebo, Then DTX401

Participants receive single peripheral IV infusion of Placebo. At week 48 eligible participants receive single peripheral IV infusion of DTX401 solution.

Group Type PLACEBO_COMPARATOR

DTX401

Intervention Type GENETIC

nonreplicating, recombinant, adeno-associated virus (AAV) serotype 8 (AAV8)

Placebo

Intervention Type OTHER

Normal Saline infusion

Oral prednisolone

Intervention Type DRUG

Participants who receive DTX401 solution will receive oral prednisolone

Placebo for oral prednisolone

Intervention Type DRUG

Participants who receive placebo will receive placebo oral prednisolone to maintain the study blind

DTX401 (Japan Only)

Participants receive single peripheral intravenous (IV)infusion of DTX401 in solution.

Group Type EXPERIMENTAL

DTX401

Intervention Type GENETIC

nonreplicating, recombinant, adeno-associated virus (AAV) serotype 8 (AAV8)

Oral prednisolone

Intervention Type DRUG

Participants who receive DTX401 solution will receive oral prednisolone

Interventions

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DTX401

nonreplicating, recombinant, adeno-associated virus (AAV) serotype 8 (AAV8)

Intervention Type GENETIC

Placebo

Normal Saline infusion

Intervention Type OTHER

Oral prednisolone

Participants who receive DTX401 solution will receive oral prednisolone

Intervention Type DRUG

Placebo for oral prednisolone

Participants who receive placebo will receive placebo oral prednisolone to maintain the study blind

Intervention Type DRUG

Other Intervention Names

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pariglasgene brecaparvovec

Eligibility Criteria

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Inclusion Criteria

* Documented GSDIa with confirmation by molecular testing or enzymatic activity on liver biopsy
* Currently receiving a therapeutic regimen of cornstarch (or equivalent), following international guidance/recommendations with stable nutrition, glycemic, and clinical status.
* Willing and able to complete the informed consent process and to comply with study procedures and visit schedule
* Females of childbearing potential and fertile males must consent to use highly effective contraception from the period following informed consent through the duration of the 144-week study and in cases of early withdrawal at least 48 weeks after the last dose of investigational product (IP). Female subjects must agree not to become pregnant. Male subjects must agree not to father a child or donate sperm

Exclusion Criteria

* Detectable pre-existing antibodies to the AAV8 capsid
* History of liver transplant, including hepatocyte cell therapy/ transplant
* History of liver disease
* Presence of liver adenoma \>5 cm in size
* Presence of liver adenoma \>3 cm and ≤5 cm in size that has a documented annual growth rate of ≥0.5 cm per year
* Significant hepatic inflammation or cirrhosis as evidenced by imaging or any of the following laboratory abnormalities: alanine aminotransferase (ALT) or aspartate aminotransferase (AST) \> upper limit of normal (ULN), total bilirubin \>1.5 × ULN, alkaline phosphatase \>2.5 × ULN
* Non-fasting triglycerides ≥1000 mg/dL
* Pregnant, breastfeeding, or planning to become pregnant (self or partner) at any time during the study.
* Current or previous participation in another gene transfer study
* History of illicit drug use within 60 days prior to screening or positive results from an 8-panel urine drug screen during the Screening Period completed at 2 time points at least 4 weeks apart
Minimum Eligible Age

8 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Ultragenyx Pharmaceutical Inc

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Medical Director

Role: STUDY_DIRECTOR

Ultragenyx Pharmaceutical Inc

Locations

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Children's Hospital of Orange County

Orange, California, United States

Site Status

Children's Hospital Colorado

Aurora, Colorado, United States

Site Status

University of Connecticut Health Center

Farmington, Connecticut, United States

Site Status

Mount Sinai

The Bronx, New York, United States

Site Status

Duke University

Durham, North Carolina, United States

Site Status

Cleveland Clinic

Cleveland, Ohio, United States

Site Status

Children's Hospital of Philadelphia

Philadelphia, Pennsylvania, United States

Site Status

University of Texas

Houston, Texas, United States

Site Status

Primary Children's Hospital

Salt Lake City, Utah, United States

Site Status

Hospital de Clinicas de Porto Alegre

Porto Alegre, Rio Grande do Sul, Brazil

Site Status

McGill University

Montreal, Quebec, Canada

Site Status

Righospitalet

Copenhagen, Capital, Denmark

Site Status

University Medical Center Eppendorf

Hamburg, , Germany

Site Status

Istituto Giannina Gaslini

Genova, Linguria, Italy

Site Status

University of Naples

Naples, , Italy

Site Status

Kumamoto University Hospital

Kumamoto, , Japan

Site Status

Osaka City General Hospital

Osaka, , Japan

Site Status

Fujita Health University Hospital

Toyoake, , Japan

Site Status

Groningen University

Groningen, , Netherlands

Site Status

Hospital Clinico Universitario de Santiago

Santiago de Compostela, A Coruna, Spain

Site Status

Countries

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United States Brazil Canada Denmark Germany Italy Japan Netherlands Spain

Related Links

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Other Identifiers

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2020-004184-12

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

2023-508750-25-00

Identifier Type: CTIS

Identifier Source: secondary_id

DTX401-CL301

Identifier Type: -

Identifier Source: org_study_id

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