A Long-Term Study of Olipudase Alfa in Patients With Acid Sphingomyelinase Deficiency

NCT ID: NCT02004704

Last Updated: 2024-07-03

Study Results

Results available

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE2

Total Enrollment

25 participants

Study Classification

INTERVENTIONAL

Study Start Date

2013-12-04

Study Completion Date

2023-09-06

Brief Summary

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The primary objective of this study was to obtain data regarding the safety of olipudase alfa in participants with acid sphingomyelinase deficiency (ASMD) who were exposed to long term treatment with olipudase alfa.

The secondary objectives of this study were to obtain data regarding the efficacy of olipudase alfa and to characterize olipudase alfa pharmacodynamics (PD) and pharmacokinetics (PK) following long-term administration.

Detailed Description

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LTS13632 is a multicenter, nonrandomized, open-label, long-term extension study of participants with ASMD who have previously participated in a study of olipudase alfa. (DFI13803 for pediatric participants and DFI13412 for adult participants).

The maximum study duration per participant was 9 years or until olipudase alfa becomes commercially accessible. Notwithstanding the above, every pediatric participant were treated in LTS13632 study for at least 3 years to comply with the requirements agreed in the olipudase alfa Pediatric Investigational Plan.

Conditions

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Sphingomyelin Lipidosis

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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GZ402665

GZ402665 administered intravenously once every 2 weeks at the dose each participant was receiving at the end of their previous olipudase alfa study, for 9 years or until olipudase alfa becomes commercially accessible, whichever comes first, unless the participant decides to enter another olipudase alfa clinical trial within the 9-year period prior to when olipudase alfa is commercially accessible.

Group Type EXPERIMENTAL

GZ402665

Intervention Type DRUG

Pharmaceutical form: Powder for concentrate for solution for infusion

Route of administration: intravenous infusion

Interventions

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GZ402665

Pharmaceutical form: Powder for concentrate for solution for infusion

Route of administration: intravenous infusion

Intervention Type DRUG

Other Intervention Names

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Olipudase alfa

Eligibility Criteria

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Inclusion Criteria

* The participant completed the treatment period of a previous study of olipudase alfa with an acceptable safety profile in the opinion of the investigator and sponsor.
* The participant and/or the participant's parent(s)/legal guardian(s) was willing and able to provide signed written informed consent.
* The participant who is female and of childbearing potential must have had a negative urine pregnancy test for beta human chorionic gonadotropin (β HCG).
* Female participants of childbearing potential and sexually mature male participants must have been willing to practice true abstinence in line with their preferred and usual lifestyle or use 2 acceptable effective methods of contraception up to 15 days following their last dose of study drug.

Exclusion Criteria

* The participant had any new condition or worsening of an existing condition which in the opinion of the investigator would make the participant unsuitable for enrollment, or could interfere with the participation or completion the study.
* The participant, in the opinion of the investigator, was unable to adhere to the requirements of the study.
* The participant was unwilling or unable to abstain from the use of alcohol for 1 day prior to and 3 days after each olipudase alfa infusion for the duration of the treatment period.
* The participant was unwilling or unable to avoid, for 10 days before and 3 days after liver biopsies, medications or herbal supplements that are potentially hepatotoxic (only participants who previously participated in the DFI13412 study).
* The participant required medication(s) that may decrease olipudase alfa activity (eg, fluoxetine, chlorpromazine; tricyclic antidepressants \[eg, imipramine, desipramine\]).

The above information was not intended to contain all considerations relevant to a participant's potential participation in a clinical trial.
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Genzyme, a Sanofi Company

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Clinical Sciences & Operations

Role: STUDY_DIRECTOR

Sanofi

Locations

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Investigational Site Number 840001

New York, New York, United States

Site Status

Investigational Site Number 056001

Leuven, , Belgium

Site Status

Investigational Site Number 076001

Porto Alegre, , Brazil

Site Status

Investigational Site Number 250002

Bron, , France

Site Status

Investigational Site Number 276002

Hochheim am Main, , Germany

Site Status

Investigational Site Number 380002

Sassari, , Italy

Site Status

Investigational Site Number 380001

Udine, , Italy

Site Status

Investigational Site Number 826001

London, , United Kingdom

Site Status

Investigational Site Number 826002

Manchester, , United Kingdom

Site Status

Countries

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United States Belgium Brazil France Germany Italy United Kingdom

References

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Scarpa M, Diaz GA, Giugliani R, Jones SA, Mengel E, Guffon N, Witters P, Ganesh J, Armstrong NM, Srivastava S, Kim Y. Long-Term Safety and Clinical Outcomes With Olipudase Alfa Enzyme Replacement Therapy in Children and Adolescents With Acid Sphingomyelinase Deficiency. J Inherit Metab Dis. 2025 Sep;48(5):e70086. doi: 10.1002/jimd.70086.

Reference Type DERIVED
PMID: 40937531 (View on PubMed)

Thurberg BL, Diaz GA, Lachmann RH, Schiano T, Wasserstein MP, Ji AJ, Zaher A, Peterschmitt MJ. Long-term efficacy of olipudase alfa in adults with acid sphingomyelinase deficiency (ASMD): Further clearance of hepatic sphingomyelin is associated with additional improvements in pro- and anti-atherogenic lipid profiles after 42 months of treatment. Mol Genet Metab. 2020 Sep-Oct;131(1-2):245-252. doi: 10.1016/j.ymgme.2020.06.010. Epub 2020 Jun 24.

Reference Type DERIVED
PMID: 32620536 (View on PubMed)

Wasserstein MP, Diaz GA, Lachmann RH, Jouvin MH, Nandy I, Ji AJ, Puga AC. Olipudase alfa for treatment of acid sphingomyelinase deficiency (ASMD): safety and efficacy in adults treated for 30 months. J Inherit Metab Dis. 2018 Sep;41(5):829-838. doi: 10.1007/s10545-017-0123-6. Epub 2018 Jan 5.

Reference Type DERIVED
PMID: 29305734 (View on PubMed)

Provided Documents

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Document Type: Study Protocol

View Document

Document Type: Statistical Analysis Plan

View Document

Other Identifiers

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2013-000051-40

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

LTS13632

Identifier Type: -

Identifier Source: org_study_id

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