Observational Study for Subjects With Pompe Disease Undergoing Immune Modulation Therapies

NCT ID: NCT01451879

Last Updated: 2017-12-08

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

Get a concise snapshot of the trial, including recruitment status, study phase, enrollment targets, and key timeline milestones.

Recruitment Status

COMPLETED

Total Enrollment

11 participants

Study Classification

OBSERVATIONAL

Study Start Date

2008-10-31

Study Completion Date

2017-10-31

Brief Summary

Review the sponsor-provided synopsis that highlights what the study is about and why it is being conducted.

Hypothesis: the effectiveness of treatment of Pompe Disease with rhGAA enzyme replacement therapy (ERT) is limited at least in part because patients develop antibodies against the provided rhGAA enzyme. Treatment with immunomodulatory drugs may dampen or eliminate the anti-rhGAA immune response in patients receiving ERT, thereby allowing for greater ERT efficacy. Studying the immune response to rhGAA may provide valuable insight into the role of the immune system in the effectiveness of ERT for Pompe Disease.

Detailed Description

Dive into the extended narrative that explains the scientific background, objectives, and procedures in greater depth.

The purpose of this research study is to determine the effect(s) of medications that alter the immune system on anti-rhGAA immune response in Pompe patients receiving rhGAA enzyme replacement therapy (ERT). The investigators would also like to determine whether treating Pompe Disease with medications that affect the immune system has any effects on the overall health or disease progression of Pompe.

Subjects will be patients between the ages of 0 months and 65 years who have been diagnosed with Pompe Disease, confirmed by mutational analysis and/or GAA enzyme activity assay.

Subjects will be eligible regardless of whether they have begun enzyme replacement therapy prior to enrollment. All Subjects will receive enzyme replacement therapy as standard of care during the course of the Study, although they may not have begun ERT treatment at the time of enrollment. In addition to ERT, subjects may receive an immunomodulatory regimen as part of their standard of care; this may include rituximab, sirolimus, methotrexate, IVIg or other immunomodulatory agents such as pharmacological chaperone N-butyldeoxynojirimycin (NB-DNJ), alone or in combination, at the discretion of their caregiver(s).

Conditions

See the medical conditions and disease areas that this research is targeting or investigating.

Pompe Disease

Keywords

Explore important study keywords that can help with search, categorization, and topic discovery.

Pompe Disease Infusion Associated Reaction Immune-modulation regimen

Study Design

Understand how the trial is structured, including allocation methods, masking strategies, primary purpose, and other design elements.

Observational Model Type

CASE_ONLY

Study Time Perspective

PROSPECTIVE

Study Groups

Review each arm or cohort in the study, along with the interventions and objectives associated with them.

Age 0 months to 65 years

Confirmed diagnosis of Pompe Disease, and clinically prescribed immune modulation regimen with agents such as rituximab, sirolimus, methotrexate, IVIg or other immunomodulatory agents such as pharmacological chaperone Miglustat, N-butyldeoxynojirimycin (NB-DNJ), alone or in combination, at the discretion of their primary/specialist caregiver.

Rituximab

Intervention Type DRUG

Clinically prescribed immune modulation regimen dosage determined by local medical provider.

Miglustat

Intervention Type DRUG

Clinically prescribed immune modulation regimen dosage determined by local medical provider.

Interventions

Learn about the drugs, procedures, or behavioral strategies being tested and how they are applied within this trial.

Rituximab

Clinically prescribed immune modulation regimen dosage determined by local medical provider.

Intervention Type DRUG

Miglustat

Clinically prescribed immune modulation regimen dosage determined by local medical provider.

Intervention Type DRUG

Other Intervention Names

Discover alternative or legacy names that may be used to describe the listed interventions across different sources.

Rituxan MabThera Zytux Zavesca N-butyldeoxynojirimaycin Mulberry extract

Eligibility Criteria

Check the participation requirements, including inclusion and exclusion rules, age limits, and whether healthy volunteers are accepted.

Inclusion Criteria

* patients between the ages of 0 months and 65 years
* diagnosed with early-onset Pompe Disease, confirmed by mutational analysis and/or GAA enzyme assay
* eligible regardless of whether they have begun enzyme replacement therapy prior to enrollment
* all subjects will receive ERT as standard of care during the course of the study, although they may not have begun ERT treatment at the time of enrollment
* subjects may receive an immunomodulatory regimen as part of their standard of care; this may include rituximab, sirolimus, methotrexate, Gamunex, Hizentra, Zavesca or other immunomodulatory agents, alone or in combination, at the discretion of their caregiver(s)

Exclusion Criteria

* subject is unable to meet the study requirements
* subjects medical condition contraindicates participation or Study Investigators feel that participation is otherwise not in the subject's best interest
* subject does not receive ERT treatment
Maximum Eligible Age

65 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

Meet the organizations funding or collaborating on the study and learn about their roles.

University of Florida

OTHER

Sponsor Role lead

Responsible Party

Identify the individual or organization who holds primary responsibility for the study information submitted to regulators.

Responsibility Role SPONSOR

Principal Investigators

Learn about the lead researchers overseeing the trial and their institutional affiliations.

Barry J Byrne, MD, PhD

Role: PRINCIPAL_INVESTIGATOR

University of Florida

Locations

Explore where the study is taking place and check the recruitment status at each participating site.

University of Florida

Gainesville, Florida, United States

Site Status

Countries

Review the countries where the study has at least one active or historical site.

United States

References

Explore related publications, articles, or registry entries linked to this study.

Elder ME, Nayak S, Collins SW, Lawson LA, Kelley JS, Herzog RW, Modica RF, Lew J, Lawrence RM, Byrne BJ. B-Cell depletion and immunomodulation before initiation of enzyme replacement therapy blocks the immune response to acid alpha-glucosidase in infantile-onset Pompe disease. J Pediatr. 2013 Sep;163(3):847-54.e1. doi: 10.1016/j.jpeds.2013.03.002. Epub 2013 Apr 16.

Reference Type RESULT
PMID: 23601496 (View on PubMed)

Other Identifiers

Review additional registry numbers or institutional identifiers associated with this trial.

IMN 439-2008

Identifier Type: OTHER

Identifier Source: secondary_id

IRB201602404

Identifier Type: -

Identifier Source: org_study_id