Treatment Effect of Tamoxifen on Patients With DMD
Primary Purpose
Duchenne Muscular Dystrophy
Status
Completed
Phase
Phase 1
Locations
Study Type
Interventional
Intervention
Tamoxifen
Sponsored by
About this trial
This is an interventional treatment trial for Duchenne Muscular Dystrophy focused on measuring Duchenne Tamoxifen
Eligibility Criteria
Inclusion Criteria:
- Ambulatory
Exclusion Criteria:
- Non Ambulatory
Sites / Locations
Arms of the Study
Arm 1
Arm Type
Other
Arm Label
open label study
Arm Description
one arm open label study
Outcomes
Primary Outcome Measures
6-minute walk distance (6MWD)
PT evaluation
Secondary Outcome Measures
North Star assesment(NSAA)
PT evaluation
Full Information
NCT ID
NCT02835079
First Posted
June 22, 2016
Last Updated
July 21, 2022
Sponsor
Hadassah Medical Organization
1. Study Identification
Unique Protocol Identification Number
NCT02835079
Brief Title
Treatment Effect of Tamoxifen on Patients With DMD
Official Title
Treatment Effect of Tamoxifen on Patients With DMD
Study Type
Interventional
2. Study Status
Record Verification Date
July 2022
Overall Recruitment Status
Completed
Study Start Date
November 2016 (Actual)
Primary Completion Date
November 2020 (Actual)
Study Completion Date
November 2020 (Actual)
3. Sponsor/Collaborators
Responsible Party, by Official Title
Principal Investigator
Name of the Sponsor
Hadassah Medical Organization
4. Oversight
Studies a U.S. FDA-regulated Drug Product
No
Studies a U.S. FDA-regulated Device Product
No
Data Monitoring Committee
No
5. Study Description
Brief Summary
Duchenne muscular dystrophy (DMD) is a progressive devastating disease that affects mainly boys, with an incidence of about 1:3,500 live births. The pathology of DMD is a result of non-repaired muscle damage that leads to muscle-tissue replacement by scar tissue, a process known as fibrosis. Currently, there is no effective treatment for the disease. The only therapy offered to these boys are steroids which slightly delayed the disease progression. The boys lose their ability to walk at around the age of 12, and die in the 4th decade of life from severe heart and lung problems.
In this study investigators will test the efficacy of Tamoxifen treatment in ambulatory DMD boys. Tamoxifen is a drug used for palliative treatment of breast cancer patients and has an outstanding safety profile. In addition, Tamoxifen was tested in the past in boys, for other pediatric indications, and showed an excellent safety with no side effects.
Tamoxifen is being tested in this study, as a therapy for DMD, for the following reasons:
(i) it was shown to have anti-fibrotic effect in multiple in-vivo systems; (ii) it assists in the repair of damaged muscles.
In other words, Tamoxifen is expected to have a synergistic effect on DMD patients, due to its dual mechanism of action. Indeed, Tamoxifen was shown to have significant beneficial effects in the mdx mouse model of DMD. Also, a small compassionate cohort of 3 boys, treated for 6 months with Tamoxifen, yielded very encouraging results.
6. Conditions and Keywords
Primary Disease or Condition Being Studied in the Trial, or the Focus of the Study
Duchenne Muscular Dystrophy
Keywords
Duchenne Tamoxifen
7. Study Design
Primary Purpose
Treatment
Study Phase
Phase 1
Interventional Study Model
Single Group Assignment
Masking
None (Open Label)
Allocation
N/A
Enrollment
19 (Actual)
8. Arms, Groups, and Interventions
Arm Title
open label study
Arm Type
Other
Arm Description
one arm open label study
Intervention Type
Drug
Intervention Name(s)
Tamoxifen
Primary Outcome Measure Information:
Title
6-minute walk distance (6MWD)
Description
PT evaluation
Time Frame
The six minute walk distance will be tested during the 36 months of the trial. For the first 12 months, the 6 minute walk test will be tested every 3 months and for the follow up period of 24 months,will be done every 6 months.
Secondary Outcome Measure Information:
Title
North Star assesment(NSAA)
Description
PT evaluation
Time Frame
The NSAA will be tested during the 36 months of the trial. For the first 12 months, the NSAA will be tested every 3 months and for the follow up period of 24 months,will be done every 6 months.
10. Eligibility
Sex
Male
Minimum Age & Unit of Time
5 Years
Maximum Age & Unit of Time
16 Years
Accepts Healthy Volunteers
No
Eligibility Criteria
Inclusion Criteria:
Ambulatory
Exclusion Criteria:
Non Ambulatory
12. IPD Sharing Statement
Citations:
PubMed Identifier
34304968
Citation
Tsabari R, Simchovitz E, Lavi E, Eliav O, Avrahami R, Ben-Sasson S, Dor T. Safety and clinical outcome of tamoxifen in Duchenne muscular dystrophy. Neuromuscul Disord. 2021 Sep;31(9):803-813. doi: 10.1016/j.nmd.2021.05.005. Epub 2021 Jun 5. Erratum In: Neuromuscul Disord. 2022 Mar;32(3):e5-e7.
Results Reference
derived
Learn more about this trial
Treatment Effect of Tamoxifen on Patients With DMD
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