NCT03643562

Brief Summary

This study was amended from expanded access to a clinical trial. Information will be collected about long-term safety and effectiveness of adrabetadex shots in the spine every 2 weeks. Participants who were already taking adrabetadex will receive their stable dose. Participants who have not ever taken it will start by receiving 400 mg. Participants will receive treatment every 2 weeks until their doctor finds it does not help them anymore, they withdraw, or the study is stopped for any reason. Participants will not receive additional study treatment after their participation in this protocol.

Trial Health

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Monitor

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
27

participants targeted

Target at below P25 for phase_3

Timeline
Completed

Started Jun 2018

Typical duration for phase_3

Geographic Reach
1 country

17 active sites

Status
terminated

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

Study Start

First participant enrolled

June 18, 2018

Completed
2 months until next milestone

First Submitted

Initial submission to the registry

August 19, 2018

Completed
4 days until next milestone

First Posted

Study publicly available on registry

August 23, 2018

Completed
3.2 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

November 2, 2021

Completed
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

November 2, 2021

Completed
2.2 years until next milestone

Results Posted

Study results publicly available

January 17, 2024

Completed
Last Updated

January 17, 2024

Status Verified

December 1, 2023

Enrollment Period

3.4 years

First QC Date

August 19, 2018

Results QC Date

November 17, 2023

Last Update Submit

December 22, 2023

Conditions

Keywords

Niemann-PickNPCNPC Type 1 (NPC1)

Outcome Measures

Primary Outcomes (1)

  • Number of Participants With Treatment-Emergent Adverse Events (TEAEs) and Serious Adverse Events (SAEs)

    An adverse event (AE) was defined as any untoward medical occurrence in a participant who received study drug without regard to possibility of causal relationship. Serious adverse events (SAEs) were defined as death, a life-threatening AE, inpatient hospitalization or prolongation of existing hospitalization, persistent or significant disability or incapacity, a congenital anomaly or birth defect, or an important medical event that jeopardized participant and required medical intervention to prevent 1 of the outcomes listed in this definition. A TEAE was defined as an AE with onset on or after the start of adrabetadex treatment. A summary of other non-serious AEs and all serious AEs, regardless of causality is located in Reported AE section.

    Baseline up to Week 134

Study Arms (1)

Adrabetadex

EXPERIMENTAL

Participants will receive prescribed adrabetadex by intra-thecal (IT) injection every 2 weeks.

Drug: Adrabetadex

Interventions

Administered via lumbar puncture (LP) and IT infusion

Also known as: VTS-270
Adrabetadex

Eligibility Criteria

Age4 Years+
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)

You may qualify if:

  • To be included in the study, a participant must meet the following criteria:
  • Is male or female and at least 4 years of age at time of screening.
  • Has a confirmed diagnosis of NPC and exhibits neurologic symptoms.
  • Has written informed consent/assent to participate.
  • Has the ability to undergo LP and IT drug administration.
  • If taking miglustat (Zavesca®), must have been on a stable dose for the past 6 weeks and be willing to remain on a stable dose for the duration of participation in the study, or discontinue miglustat use at least 6 weeks before entry into the study (Day 1).
  • If has a history of seizures, the condition is adequately controlled as per protocol requirements.
  • Agrees to discontinue any investigational treatments (other than adrabetadex) for at least 1 month before first dose on Day 1.
  • If engaging in heterosexual sex, agrees to use a protocol-defined method of contraception throughout the study, and until 30 days after completing the study.
  • Has a responsible adult who the investigator determines is able and willing to comply with study requirements and a parent/guardian who will accompany the participant to study visits.

You may not qualify if:

  • Participants will be excluded from the study if they meet any of the following criteria:
  • Weighs less than 15 kg.
  • Has a history of hypersensitivity reactions to any product containing 2-hydroxypropyl-β-cyclodextrin (HP-β-CD) or has a history of hypersensitivity reactions or allergy to anesthesia/sedation.
  • Has received treatment with any investigational product (other than adrabetadex) within 1 month prior to Day 1 of treatment.
  • Is pregnant or nursing.
  • Has systemic infection or uncontrolled psychosis.
  • Has known history of a bleeding disorder.
  • Has used anticoagulants within 2 months of entry into the study.
  • Per protocol, or in the opinion of the investigator:
  • has laboratory values that would preclude participation
  • has suspected infection of the central nervous system (CNS)
  • has a spinal deformity that could impact performance of repeated LPs
  • has a serious skin infection in the lumbar region or evidence of obstructive or normal pressure hydrocephalus
  • is unable to comply with the study requirements
  • has a medical condition that might increase the risk of participation

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (17)

University of Arkansas System

Little Rock, Arkansas, 72205, United States

Location

Loma Linda University Health System

Loma Linda, California, 92354, United States

Location

Children's Hospital Los Angeles

Los Angeles, California, 90027, United States

Location

Children's Hospital of Orange County

Orange, California, 92868, United States

Location

Children's Hospital Colorado

Aurora, Colorado, 80045, United States

Location

Children's National Hospital

Washington D.C., District of Columbia, 20010, United States

Location

Rare Disease Research, LLC

Atlanta, Georgia, 30318, United States

Location

Rush University Medical Center

Chicago, Illinois, 60612, United States

Location

Gillette Children's Specialty Healthcare

Saint Paul, Minnesota, 55101, United States

Location

Children's Specialty Center of Nevada

Las Vegas, Nevada, 89109, United States

Location

NYU Langone Medical Center

New York, New York, 10017, United States

Location

Cincinnati Children's Hospital Medical Center

Cincinnati, Ohio, 45229, United States

Location

Oregon Health & Science University

Portland, Oregon, 97239, United States

Location

The Children's Hospital at TriStar Centennial

Nashville, Tennessee, 37203, United States

Location

Vanderbilt University Medical Center

Nashville, Tennessee, 37232, United States

Location

Dell Children's Medical Center of Central Texas

Austin, Texas, 78723, United States

Location

Carilion Medical Center, Carilion Clinic

Roanoke, Virginia, 24014, United States

Location

MeSH Terms

Conditions

Niemann-Pick Disease, Type C

Interventions

2-Hydroxypropyl-beta-cyclodextrin

Condition Hierarchy (Ancestors)

Niemann-Pick DiseasesSphingolipidosesLysosomal Storage Diseases, Nervous SystemBrain Diseases, Metabolic, InbornBrain Diseases, MetabolicBrain DiseasesCentral Nervous System DiseasesNervous System DiseasesHistiocytosis, Non-Langerhans-CellHistiocytosisLymphatic DiseasesHemic and Lymphatic DiseasesMetabolism, Inborn ErrorsGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesLipidosesLipid Metabolism, Inborn ErrorsLysosomal Storage DiseasesMetabolic DiseasesNutritional and Metabolic DiseasesLipid Metabolism Disorders

Intervention Hierarchy (Ancestors)

beta-CyclodextrinsCyclodextrinsMacrocyclic CompoundsPolycyclic CompoundsDextrinsStarchDietary CarbohydratesCarbohydratesGlucansPolysaccharides

Limitations and Caveats

This study was terminated early when the Sponsor had determined that the benefit-risk profile of adrabetadex was not favorable and the clinical development program for adrabetadex was discontinued.

Results Point of Contact

Title
Executive Vice President, Regulatory Affairs
Organization
Mandos, LLC

Study Officials

  • Clinical Study Lead

    Mandos LLC

    STUDY DIRECTOR

Publication Agreements

PI is Sponsor Employee
No
Restrictive Agreement
Yes

Study Design

Study Type
interventional
Phase
phase 3
Allocation
NA
Masking
NONE
Purpose
TREATMENT
Intervention Model
SINGLE GROUP
Model Details: Continued treatment for children at least 4 years of age
Sponsor Type
INDUSTRY
Responsible Party
SPONSOR

Study Record Dates

First Submitted

August 19, 2018

First Posted

August 23, 2018

Study Start

June 18, 2018

Primary Completion

November 2, 2021

Study Completion

November 2, 2021

Last Updated

January 17, 2024

Results First Posted

January 17, 2024

Record last verified: 2023-12

Data Sharing

IPD Sharing
Will not share

Locations