Long-term Safety and Efficacy of Odevixibat in Patients With Alagille Syndrome

NCT ID: NCT05035030

Last Updated: 2025-12-30

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

PHASE3

Total Enrollment

70 participants

Study Classification

INTERVENTIONAL

Study Start Date

2021-09-03

Study Completion Date

2026-12-31

Brief Summary

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The purpose of this study is to assess the long-term safety and effectiveness of odevixibat in participants with Alagille syndrome (ALGS).

The participants of this study will have ALGS a rare genetic disorder that can affect multiple organ systems of the body including the liver, heart, skeleton, eyes and kidneys. Common symptoms, which often develop during the first three months of life, include blockage of the flow of bile from the liver (cholestasis), yellowing of the skin and mucous membranes (jaundice), poor weight gain and growth and severe itching (pruritis).

The drug used for the study is odevixibat and was authorized for the treatment of cholestatic pruritus in infants with ALGS over 12 months of age by the United States Food and Drug Administration on 13 June 2023.

Detailed Description

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This Phase 3, open-label, multi-center extension study will have two groups of participants: Cohort 1 (participants who participated in Study A4250-012 \[NCT04674761; ASSERT\] and meet the entry criteria for this study) and Cohort 2 (infants under 12 months of age) with ALGS.

The study will consist of 2 or 3 periods:

1. A 'Treatment period' of 72 weeks (cohort 1) or 12 weeks (cohort 2). Participants will visit the clinic every 4 to 12 weeks and will receive a dose of 120 μg/kg odevixibat daily.
2. An 'Optional extension period' where participants who wish to continue receiving odevixibat after the 'treatment period' will have the opportunity to remain on treatment with visits every 16 weeks until the drug is commercially available. The optional extension is available provided continued use is supported by the risk-benefit profile, the participant has not been previously withdrawn or discontinued from the study, and the study is not terminated by the Sponsor.
3. A 'Safety follow-up period' of 4 weeks (cohort 1) or 2 weeks (cohort 2). The Safety Follow-up Period will not occur for those who remain on treatment in the optional extension period.

Participants will need to complete an e-diary and questionnaires throughout the study (cohort 1 only). Participants will undergo blood samplings, urine collections (cohort 1 only), physical examinations, and clinical evaluations. They may continue some other medications, but the details need to be recorded.

Conditions

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Alagille Syndrome

Study Design

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Allocation Method

NA

Intervention Model

SINGLE_GROUP

Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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Odevixibat (A4250)

Capsules for oral administration once daily for 72 weeks.

Group Type EXPERIMENTAL

Odevixibat

Intervention Type DRUG

Odevixibat is a small molecule and selective inhibitor of IBAT.

Interventions

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Odevixibat

Odevixibat is a small molecule and selective inhibitor of IBAT.

Intervention Type DRUG

Other Intervention Names

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A4250

Eligibility Criteria

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Inclusion Criteria

Cohort 1 :

1. Completion of the 24-week Treatment Period of Study A4250-012
2. Signed informed consent and assent as appropriate. Patients who turn 18 years of age (or legal age per country) during the study will be required to re-consent to remain on the study
3. Caregivers (and age-appropriate patients) must be willing and able to use an electronic diary (eDiary) device as required by the study
4. Sexually active males and females must agree to use a reliable contraceptive method with ≤1% failure rate (such as hormonal contraception, intra-uterine device, or complete abstinence) from signed informed consent through 90 days after last dose of study drug.

Cohort 2 :

1. Infant with clinically confirmed ALGS , ≤11 months of age at Study Day 1
2. Body weight ≥2 kg at Study Day 1
3. Gestational age ≥36 weeks. For children born with gestational age between 32 and 36 weeks, a postmenstrual age of ≥36 weeks is required .
4. Signed parent/legal guardian informed consent.

Exclusion Criteria

Cohort 1 :

1. Decompensated liver disease, history or presence of clinically significant ascites, variceal hemorrhage, and/or encephalopathy
2. Patients who were not compliant with study drug treatment or procedures in Study A4250-012
3. Any other conditions or abnormalities which, in the opinion of the investigator, may compromise the safety of the patient, or interfere with the patient participating in or completing the study
4. Known hypersensitivity to any components of odevixibat

Cohort 2 :

1. Patient with past medical history or ongoing presence of other types of liver disease including, but not limited to, the following:

1. Biliary atresia of any kind
2. Progressive familial intrahepatic cholestasis (PFIC)
3. Benign recurrent intrahepatic cholestasis
2. Patient with a past medical history or ongoing presence of any other disease or condition known to interfere with the absorption, distribution, metabolism (specifically bile acid metabolism), or excretion of drugs in the intestine, including but not limited to, inflammatory bowel disease
3. Patient with past medical history or ongoing chronic diarrhea requiring intravenous fluid or nutritional intervention for treatment of the diarrhea and/or its sequelae
4. Patient has a confirmed past diagnosis of infection with human immunodeficiency virus or other present and active, clinically significant chronic infection
5. Recent infection requiring hospitalization or treatment with parenteral anti-infective within 4 weeks of Study Day 1 or completion of oral anti-infective treatment within 2 weeks prior to the Screening Visit
6. Cancer diagnosis (except for basal cell carcinoma)
7. Chronic kidney disease with an impaired renal function and a glomerular filtration rate \<70 mL/min/1.73 m2
8. Patient with surgical history of disruption of the enterohepatic circulation (biliary diversion surgery) within 6 months prior to the Screening Visit
9. Patient has had a liver transplant, or a liver transplant is planned within 6 months of Study Day 1
10. Decompensated liver disease, history or presence of clinically significant ascites, variceal hemorrhage, and/or encephalopathy
11. International normalized ratio (INR) \>1.4 (the patient may be treated with Vitamin K, and if INR is ≤1.4 at resampling the patient may be enrolled)
12. Serum alanine aminotransferase (ALT) \>10 × upper limit of normal (ULN) at Screening
13. Serum ALT \>15 × ULN at any time point during the last 6 months unless an alternate etiology was confirmed for the elevation
14. Total bilirubin \>15 × ULN at Screening
15. Patient suffers from uncontrolled, recalcitrant pruritic condition other than ALGS. Examples include, but not limited to, refractory atopic dermatitis or other primary pruritic skin diseases.
16. Patient exposed to alcohol or substance abuse in utero
17. Bile acid or lipid binding resins and medications that slow gastrointestinal motility
18. Patient has had investigational exposure to a drug, biologic agent, or medical device within 30 days prior to the Screening Visit, or 5 half-lives of the study agent, whichever is longer
19. Any other conditions or abnormalities which, in the opinion of the investigator may compromise the safety of the patient, or interfere with the patient participating in or completing the study
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Albireo, an Ipsen Company

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Ipsen Medical Director

Role: STUDY_DIRECTOR

Ipsen

Locations

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Rady Children's Hospital

San Diego, California, United States

Site Status RECRUITING

UCSF

San Francisco, California, United States

Site Status ACTIVE_NOT_RECRUITING

University of California San Francisco (UCSF)

San Francisco, California, United States

Site Status RECRUITING

Children's Healthcare of Atlanta

Atlanta, Georgia, United States

Site Status RECRUITING

Riley Hospital for Children at IU Health

Indianapolis, Indiana, United States

Site Status RECRUITING

Johns Hopkins Hospital

Baltimore, Maryland, United States

Site Status RECRUITING

Boston Children's Hospital

Boston, Massachusetts, United States

Site Status COMPLETED

Children's Mercy Hospital and Clinics

Kansas City, Missouri, United States

Site Status RECRUITING

Northwell Health System

New Hyde Park, New York, United States

Site Status NOT_YET_RECRUITING

Hassenfeld Children's Hospital at NYU Langone

New York, New York, United States

Site Status RECRUITING

The Childrens Hospital at Montefiore Albert Einstein School of Medicine

The Bronx, New York, United States

Site Status ACTIVE_NOT_RECRUITING

Atrium Health Carolinas Medical

Durham, North Carolina, United States

Site Status RECRUITING

Cincinnati Children's Hospital

Cincinnati, Ohio, United States

Site Status NOT_YET_RECRUITING

Oregon Health Science University School of Medicine

Portland, Oregon, United States

Site Status RECRUITING

Monroe Carell Jr. Childrens Hospital at Vanderbilt

Nashville, Tennessee, United States

Site Status RECRUITING

Childrens Medical Center of Dallas University of Texas Southwestern

Dallas, Texas, United States

Site Status RECRUITING

Texas Children's Hospital

Houston, Texas, United States

Site Status RECRUITING

Texas Liver Institute

San Antonio, Texas, United States

Site Status RECRUITING

Cliniques Universitaires Saint-Luc Bruxelles

Brussels, , Belgium

Site Status ACTIVE_NOT_RECRUITING

Hôpital Femme Mère Enfant de Lyon

Bron, , France

Site Status WITHDRAWN

Antenne pediatrique du CIC-Hopital Jeanne De Flandre

Lille, , France

Site Status ACTIVE_NOT_RECRUITING

Hopital Necker Enfants Malades

Paris, , France

Site Status ACTIVE_NOT_RECRUITING

Charité - Universitätsmedizin Berlin

Berlin, , Germany

Site Status ACTIVE_NOT_RECRUITING

Medizinische Hochschul

Hanover, , Germany

Site Status ACTIVE_NOT_RECRUITING

Universitatsklinik fur Kinder-und Jugendmedizin Tubingen

Tübingen, , Germany

Site Status ACTIVE_NOT_RECRUITING

AOU Meyer

Florence, , Italy

Site Status ACTIVE_NOT_RECRUITING

Azienda Ospedale University

Padua, , Italy

Site Status ACTIVE_NOT_RECRUITING

Ospedale Pediatrico Bambino Gesu

Rome, , Italy

Site Status ACTIVE_NOT_RECRUITING

University of Malaya Medical Center

Kuala Lumpur, , Malaysia

Site Status RECRUITING

Universitair Medisch Centrum Groningen

Groningen, , Netherlands

Site Status COMPLETED

Wilhelmina Children's Hospital UMCU Utrecht

Utrecht, , Netherlands

Site Status ACTIVE_NOT_RECRUITING

Instytut Pomnik-Centrum Zdrowia Dzieck

Warsaw, , Poland

Site Status ACTIVE_NOT_RECRUITING

Istanbul University Istanbul Medical Faculty Hospital

Istanbul, , Turkey (Türkiye)

Site Status ACTIVE_NOT_RECRUITING

Birmingham Women's and Children's NHS Foundation Trust

Birmingham, , United Kingdom

Site Status NOT_YET_RECRUITING

King's College Hospital NHS Foundation Trust King's College Hospital Paediatric Research

London, , United Kingdom

Site Status RECRUITING

Countries

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United States Belgium France Germany Italy Malaysia Netherlands Poland Turkey (Türkiye) United Kingdom

Central Contacts

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Ipsen Recruitment enquiries

Role: CONTACT

Phone: See e mail

Email: [email protected]

Facility Contacts

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Trung Van

Role: primary

Ashley Thomas

Role: backup

Heather Wasserkbury

Role: primary

Rozanne Groen

Role: primary

Yee Siong Chaw

Role: primary

Michelle Tran

Role: primary

Other Identifiers

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2023-509028-17-00

Identifier Type: CTIS

Identifier Source: secondary_id

2021-000996-36

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

A4250-015

Identifier Type: -

Identifier Source: org_study_id