The Study of an Investigational Drug, Revusiran (ALN-TTRSC), for the Treatment of Transthyretin (TTR)-Mediated Amyloidosis in Patients Whose Disease Has Continued to Worsen Following Liver Transplant
Primary Purpose
Transthyretin (TTR)-Mediated Amyloidosis, Familial Amyloidotic Polyneuropathy (FAP), ATTR Amyloidosis
Status
Completed
Phase
Phase 2
Locations
International
Study Type
Interventional
Intervention
Revusiran
Sponsored by
About this trial
This is an interventional treatment trial for Transthyretin (TTR)-Mediated Amyloidosis focused on measuring FAP, Familial Amyloidotic Polyneuropathy, ATTR Amyloidosis, Orthotopic Liver Transplant, RNAi therapeutic
Eligibility Criteria
Inclusion Criteria:
- Diagnosis of FAP (familial amyloidotic polyneuropathy) with documented TTR mutation
- Received an orthotopic liver transplant ≥12 months before the date of informed consent
- An increase in polyneuropathy disability (PND) score post-transplant
- Polyneuropathy Disability score of ≤3b
Exclusion Criteria:
- New York Heart Association (NYHA) classification of >2
- Other known causes of sensorimotor or autonomic neuropathy (eg, autoimmune disease)
Sites / Locations
- Clinical Trial SIte
- Clinical Trial SIte
- Clinical Trial SIte
- Clinical Trial SIte
- Clinical Trial Site
- Clinical Trial SIte
Arms of the Study
Arm 1
Arm Type
Experimental
Arm Label
All Patients
Arm Description
All patients who received at least 1 dose of revusiran (ALN-TTRSC)
Outcomes
Primary Outcome Measures
Percentage Change From Baseline in Serum TTR at Month 6
A negative percentage change from baseline at Month 6 indicates a reduction in serum TTR level.
Secondary Outcome Measures
Percentage Change From Baseline in Serum TTR Over 18 Months
A negative percentage change from baseline indicates a reduction in serum TTR level.
Change From Baseline in Modified Neurological Impairment Score (mNIS +7) Composite Score Over 18 Months
The mNIS+7 is a composite score that measures neurologic impairment which includes the following components: physical exam of lower limbs, upper limbs and cranial nerves to assess motor strength/weakness, electrophysiologic measurement of small and large nerve fiber function, sensory testing and postural blood pressure. The minimum and maximum values are 0 and 304, respectively. A higher score indicates a worse outcome. A negative change from baseline indicates an improvement.
Norfolk Quality of Life-Diabetic Neuropathy (QoL-DN) Questionnaire Score
The Norfolk QoL-DN questionnaire is a standardized 35-item patient-reported outcomes measure that is sensitive to the different features of diabetic neuropathy - small fiber, large fiber, and autonomic nerve function. The minimum and maximum values are -4 and 136, respectively. A higher score indicates a worse outcome.
Number of Participants in Each Polyneuropathy Disability (PND) Stage Based on Worst Post-Baseline Score
PND Scores: Stage 0: No symptoms, Stage 1: Sensory disturbances but preserved walking capabilities, Stage 2: Impaired walking capacity, but ability to walk without a stick or crutches, Stage 3A/B: Walking with help of 1 or 2 sticks or crutches, Stage 4: confined to wheel chair. For each stage (0-4) at baseline, the number of participants is presented at their worst post-baseline score for each stage (0-4) post-baseline. Worst post-baseline is defined the highest PND classification for a participant recorded after the first dose of study drug.
Full Information
NCT ID
NCT02595983
First Posted
October 30, 2015
Last Updated
March 26, 2019
Sponsor
Alnylam Pharmaceuticals
1. Study Identification
Unique Protocol Identification Number
NCT02595983
Brief Title
The Study of an Investigational Drug, Revusiran (ALN-TTRSC), for the Treatment of Transthyretin (TTR)-Mediated Amyloidosis in Patients Whose Disease Has Continued to Worsen Following Liver Transplant
Official Title
An Open-label Study to Evaluate the Efficacy and Safety of Revusiran in Patients With Transthyretin-mediated Familial Amyloidotic Polyneuropathy With Disease Progression Post-Orthotopic Liver Transplant
Study Type
Interventional
2. Study Status
Record Verification Date
March 2019
Overall Recruitment Status
Completed
Study Start Date
October 2015 (undefined)
Primary Completion Date
February 6, 2017 (Actual)
Study Completion Date
February 6, 2017 (Actual)
3. Sponsor/Collaborators
Responsible Party, by Official Title
Sponsor
Name of the Sponsor
Alnylam Pharmaceuticals
4. Oversight
Data Monitoring Committee
Yes
5. Study Description
Brief Summary
The purpose of this study was to evaluate the safety and effectiveness of revusiran (ALN-TTRSC) in adults with transthyretin-mediated amyloidosis (ATTR), whose disease has continued to worsen after liver transplantation. Dosing has been discontinued; patients are being followed-up for safety.
6. Conditions and Keywords
Primary Disease or Condition Being Studied in the Trial, or the Focus of the Study
Transthyretin (TTR)-Mediated Amyloidosis, Familial Amyloidotic Polyneuropathy (FAP), ATTR Amyloidosis, Familial Amyloid Neuropathies
Keywords
FAP, Familial Amyloidotic Polyneuropathy, ATTR Amyloidosis, Orthotopic Liver Transplant, RNAi therapeutic
7. Study Design
Primary Purpose
Treatment
Study Phase
Phase 2
Interventional Study Model
Single Group Assignment
Masking
None (Open Label)
Allocation
N/A
Enrollment
12 (Actual)
8. Arms, Groups, and Interventions
Arm Title
All Patients
Arm Type
Experimental
Arm Description
All patients who received at least 1 dose of revusiran (ALN-TTRSC)
Intervention Type
Drug
Intervention Name(s)
Revusiran
Other Intervention Name(s)
ALN-TTRSC
Intervention Description
500mg Revusiran by subcutaneous (sc) injection
Primary Outcome Measure Information:
Title
Percentage Change From Baseline in Serum TTR at Month 6
Description
A negative percentage change from baseline at Month 6 indicates a reduction in serum TTR level.
Time Frame
Month 6
Secondary Outcome Measure Information:
Title
Percentage Change From Baseline in Serum TTR Over 18 Months
Description
A negative percentage change from baseline indicates a reduction in serum TTR level.
Time Frame
Weeks 3, 7, 12, 18, 24, 26 (Month 6), 39 (Month 9), 52 (Month 12), 57, 78 (Month 18)
Title
Change From Baseline in Modified Neurological Impairment Score (mNIS +7) Composite Score Over 18 Months
Description
The mNIS+7 is a composite score that measures neurologic impairment which includes the following components: physical exam of lower limbs, upper limbs and cranial nerves to assess motor strength/weakness, electrophysiologic measurement of small and large nerve fiber function, sensory testing and postural blood pressure. The minimum and maximum values are 0 and 304, respectively. A higher score indicates a worse outcome. A negative change from baseline indicates an improvement.
Time Frame
Baseline, Months 6, 12, 18
Title
Norfolk Quality of Life-Diabetic Neuropathy (QoL-DN) Questionnaire Score
Description
The Norfolk QoL-DN questionnaire is a standardized 35-item patient-reported outcomes measure that is sensitive to the different features of diabetic neuropathy - small fiber, large fiber, and autonomic nerve function. The minimum and maximum values are -4 and 136, respectively. A higher score indicates a worse outcome.
Time Frame
Baseline, Months 6, 12, 18
Title
Number of Participants in Each Polyneuropathy Disability (PND) Stage Based on Worst Post-Baseline Score
Description
PND Scores: Stage 0: No symptoms, Stage 1: Sensory disturbances but preserved walking capabilities, Stage 2: Impaired walking capacity, but ability to walk without a stick or crutches, Stage 3A/B: Walking with help of 1 or 2 sticks or crutches, Stage 4: confined to wheel chair. For each stage (0-4) at baseline, the number of participants is presented at their worst post-baseline score for each stage (0-4) post-baseline. Worst post-baseline is defined the highest PND classification for a participant recorded after the first dose of study drug.
Time Frame
Baseline, Months 6, 12, 18
10. Eligibility
Sex
All
Minimum Age & Unit of Time
18 Years
Accepts Healthy Volunteers
No
Eligibility Criteria
Inclusion Criteria:
Diagnosis of FAP (familial amyloidotic polyneuropathy) with documented TTR mutation
Received an orthotopic liver transplant ≥12 months before the date of informed consent
An increase in polyneuropathy disability (PND) score post-transplant
Polyneuropathy Disability score of ≤3b
Exclusion Criteria:
New York Heart Association (NYHA) classification of >2
Other known causes of sensorimotor or autonomic neuropathy (eg, autoimmune disease)
Overall Study Officials:
First Name & Middle Initial & Last Name & Degree
John Vest, MD
Organizational Affiliation
Alnylam Pharmaceuticals
Official's Role
Study Director
Facility Information:
Facility Name
Clinical Trial SIte
City
Paris
Country
France
Facility Name
Clinical Trial SIte
City
Münster
Country
Germany
Facility Name
Clinical Trial SIte
City
Porto
Country
Portugal
Facility Name
Clinical Trial SIte
City
Majorca
Country
Spain
Facility Name
Clinical Trial Site
City
Umea
Country
Sweden
Facility Name
Clinical Trial SIte
City
London
Country
United Kingdom
12. IPD Sharing Statement
Learn more about this trial
The Study of an Investigational Drug, Revusiran (ALN-TTRSC), for the Treatment of Transthyretin (TTR)-Mediated Amyloidosis in Patients Whose Disease Has Continued to Worsen Following Liver Transplant
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