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A Study to Assess the Long-Term Safety and Efficacy of Eplontersen (Formerly Known as ION-682884, IONIS-TTR-LRx and AKCEA-TTR-LRx) in Patients With Hereditary Transthyretin-Mediated Amyloid Polyneuropathy

Phase 3
Active, not recruiting
Conditions
Hereditary Transthyretin-Mediated Amyloid Polyneuropathy
Interventions
Registration Number
NCT05071300
Lead Sponsor
Ionis Pharmaceuticals, Inc.
Brief Summary

The purpose of this study is to evaluate the safety and tolerability of extended dosing with Eplontersen in participants with hereditary transthyretin-mediated amyloid polyneuropathy (hATTR-PN).

Detailed Description

This is a multicenter, open-label, Phase 3 study in up to approximately 140 participants. Eligible participants will receive Eplontersen once every 4 weeks for up to 157 weeks. Participants will also receive daily supplemental doses of the recommended daily allowance (RDA) of vitamin A. This study will consist of the following periods: less than or equal to (≤) 8-week screening period, a 157 weeks treatment period, and a 24-week post-treatment evaluation period.

Recruitment & Eligibility

Status
ACTIVE_NOT_RECRUITING
Sex
All
Target Recruitment
140
Inclusion Criteria
  1. Satisfactory completion of ION-682884-CS3 (NCT04136184) (Index Study) as judged by the Investigator and Sponsor, or diagnosis of hATTR-PN and satisfactory completion of either study ISIS 420915-CS101 or study 2018-P001436 (NCT03702829) (both are Investigator-Sponsored studies with inotersen - the unconjugated version of Eplontersen) as judged by the Investigator and Sponsor.

  2. Must have given written informed consent (signed and dated) and any authorizations required by local law and be able to comply with all study requirements.

  3. Satisfy the following:

    1. Females: must be non-pregnant and non-lactating and either:

      • Surgically sterile (e.g., tubal occlusion, hysterectomy, bilateral salpingectomy, bilateral oophorectomy);
      • Post-menopausal (defined as 12 months of spontaneous amenorrhea in females > 55 years of age or, in females ≤ 55 years, 12 months of spontaneous amenorrhea without an alternative medical cause and follicle-stimulating hormone (FSH) levels in the postmenopausal range for the laboratory involved;
      • Abstinent*;
      • If engaged in sexual relations of child-bearing potential, agree to use highly effective contraceptive methods from the time of signing the informed consent form until at least 24 weeks after the last dose of Eplontersen and agree to receive pregnancy tests per protocol.
    2. Males: Surgically sterile (i.e., bilateral orchidectomy) or abstinent*, if engaged in sexual relations with a woman of child-bearing potential (WOCBP), the participant or the participant's non-pregnant female partner must use a highly effective contraceptive method from the time of signing the informed consent form until at least 24 weeks after the last dose of Eplontersen. *Abstinence (i.e., refraining from heterosexual intercourse throughout the duration of study participation) is only acceptable as true abstinence, i.e., when this is in line with the preferred and usual lifestyle of the participant. Periodic abstinence (e.g., calendar, ovulation, symptothermal, post-ovulation methods), declaration of abstinence for the duration of a trial and withdrawal are not acceptable methods of contraception.

  4. Willingness to adhere to vitamin A supplementation per protocol.

Exclusion Criteria
  1. Have any new condition or worsening of existing condition that in the opinion of the Investigator or Sponsor would make the participant unsuitable for enrollment or could interfere with the participant taking part in or completing the study.

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
EplontersenEplontersenEplontersen will be administered by subcutaneous (SC) injection once every 4 weeks for up to 3 years (157 weeks).
Primary Outcome Measures
NameTimeMethod
Change From Baseline in Platelet CountBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Renal FunctionBaseline to Week 181
Number of Participants with Clinically Significant Changes from Baseline in TransaminasesBaseline to Week 181
Change From Baseline in Adverse EventsBaseline to Week 181
Change From Baseline in Number of Concomitant Medications UsedBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Vital SignsBaseline to Week 181
Change From Baseline in Body WeightBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Physical Examination FindingsBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Clinical Laboratory TestsBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Electrocardiogram (ECG) ParametersBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Thyroid Panel TestsBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Coagulation TestsBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Inflammatory Panel TestsBaseline to Week 181
Number of Participants With Clinically Significant Changes From Baseline in Complement and Immunogenicity TestsBaseline to Week 181
Secondary Outcome Measures
NameTimeMethod
Change From Baseline in Neuropathy Symptom and Change Score (NSC)Baseline to Week 181

NSC score is a questionnaire composed of 38 questions that assess the presence and severity of neuropathy symptoms (including weakness, loss of temperature and pain sensation, and manifestations associated with autonomic nervous system dysfunction).

Change From Baseline in Neuropathy Impairment Score (NIS)Baseline to Week 181

NIS is a composite, quantitative measure of both large-and small-fiber dysfunction used to evaluate the participant's muscle strength, sensation, and reflexes. Total NIS is graded on a scale of 0-244, with a higher score indicating greater impairment.

Change From Baseline in Physical Component Summary Score (PCS) of 36-Item Short Form Survey (SF-36)Baseline to Week 181

The SF-36 is composed of 8 multi-item scales (35 items) assessing physical function (10 items), role limitations due to physical health problems (4 items), bodily pain (2 items), general health (5 items), vitality (4 items), social functioning (2 items), role limitations due to emotional problems (3 items) and emotional well-being (5 items). Each of the 8 scales is scored from 0 to 100 with higher scores indicating better health. The 8 scales can be aggregated into a PCS score, which is also scaled from 0 to 100 with higher scores indicating better health.

Change From Baseline in Polyneuropathy Disability Score (PND)Baseline to Week 181

PND score assesses disease severity using a 5-stage scoring system. It includes Stage 0: no impairment; 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 or bedridden.

Change From Baseline in Modified Body Mass Index (mBMI)Baseline to Week 181

mBMI is defined as body mass index in kilograms per square meter (kg/m\^2) multiplied by serum albumin in grams per liter (g/L).

Change From Baseline in Norfolk Quality of Life-Diabetic Neuropathy (Norfolk QOL-DN) QuestionnaireBaseline to Week 181

The Norfolk QoL-DN score is a measure of physical function/large fiber neuropathy, symptoms, activities of daily living, small fiber neuropathy, and autonomic neuropathy. The Norfolk QoL-DN total score has a range of -4 to 136, and a higher score indicates poorer quality of life.

Change From Baseline in Serum Transthyretin (TTR) ConcentrationBaseline to Week 181
Change From Baseline in Composite Autonomic Symptom Score-31 (COMPASS-31)Baseline to Week 181

COMPASS-31 is a 31-question participant-reported assessment that measures autonomic symptoms across 6 weighted domains on a 100-point scale: orthostatic intolerance (40 points), vasomotor (5 points), secretomotor (15 points), gastrointestinal (25 points), bladder (10 points), and pupillomotor (15 points). A higher score indicates worse autonomic dysfunction.

Change From Baseline in 5 Level EQ-5D (EQ-5D-5L)Baseline to Week 181

The EQ-5D-5L is a standard measure of health-related quality of life. EQ-5D-5L consists of two components: a health state profile and a visual analog scale (VAS). EQ-5D health state profile comprises 5 dimensions: mobility, self-care, usual activities, pain/discomfort, and anxiety/depression. Each dimension has 5 levels: 1=no problems, 2=slight problems, 3=moderate problems, 4=severe problems, and 5=extreme problems. The 5D-5L systems are converted into a single index utility score between 0 to 1, where a higher score indicates a better health state. The VAS records the participant's health on a 0-100 mm VAS scale, with 0 indicating "the worst health you can imagine" and 100 indicating "the best health you can imagine". Higher scores of EQ VAS indicate better health.

Trial Locations

Locations (33)

Hôpital de la Timone

🇫🇷

Marseille, France

Hospital Clínico San Carlos

🇪🇸

Madrid, Spain

Hospital Son Llàtzer

🇪🇸

Palma De Mallorca, Spain

Norrlands Universitetssjukhus

🇸🇪

Umeå, Sweden

Mayo Clinic

🇺🇸

Scottsdale, Arizona, United States

Indiana University Health University Hospital

🇺🇸

Indianapolis, Indiana, United States

Johns Hopkins University Neurology Research Office

🇺🇸

Baltimore, Maryland, United States

Boston University School of Medicine

🇺🇸

Boston, Massachusetts, United States

The Neurological Institute of New York

🇺🇸

New York, New York, United States

University of North Carolina Hospitals - Neurology Clinic

🇺🇸

Chapel Hill, North Carolina, United States

Oregon Health and Science University

🇺🇸

Portland, Oregon, United States

University of Washington Medical Center

🇺🇸

Seattle, Washington, United States

Hospital Italiano de Buenos Aires

🇦🇷

Buenos Aires, Argentina

Instituto Fleni

🇦🇷

Buenos Aires, Argentina

Hospital El Cruce

🇦🇷

Florencio Varela, Argentina

Perron Institute for Neurological and Translational Science

🇦🇺

Murdoch, Western Australia, Australia

Hospital Universitario Clementino Fraga Filho

🇧🇷

Botafogo, Rio De Janeiro, Brazil

Universidade Estadual de Campinas

🇧🇷

Campinas, Brazil

Instituto de Neurologia de Curitiba

🇧🇷

Curitiba, Brazil

Hospital das Clínicas da Faculdade de Medicina de Ribeirão Preto

🇧🇷

São Paulo, Brazil

Vancouver General Hospital

🇨🇦

Vancouver, British Columbia, Canada

Toronto General Hospital

🇨🇦

Toronto, Ontario, Canada

The Cyprus Institute of Neurology and Genetics

🇨🇾

Égkomi, Cyprus

Centre Hospitalier Universitaire de Toulouse

🇫🇷

Toulouse, Haute-Garonne, France

Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) - Fondazione Istituto Neurologico Carlo Besta

🇮🇹

Milano, Italy

Fondazione IRCCS Policlinico San Matteo

🇮🇹

Pavia, Italy

Centro Hospitalar Universitário Lisboa Norte - Hospital De Santa Maria

🇵🇹

Lisboa, Portugal

Centro Hospitalar Universitário do Porto - Hospital Geral de Santo Antonio

🇵🇹

Porto, Portugal

China Medical University Hospital

🇨🇳

Taichung, Taiwan

National Taiwan University Hospital

🇨🇳

Taipei City, Taiwan

Taipei Veterans General Hospital

🇨🇳

Taipei, Taiwan

Chang Gung Memorial Hospital

🇨🇳

Taoyuan, Taiwan

Istanbul Üniversitesi - Istanbul Tip Fakültesi

🇹🇷

Istanbul, Turkey

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