NEURO-TTRansform: A Study to Evaluate the Efficacy and Safety of Eplontersen (Formerly Known as ION-682884, IONIS-TTR-LRx and AKCEA-TTR-LRx) in Participants With Hereditary Transthyretin-Mediated Amyloid Polyneuropathy

NCT ID: NCT04136184

Last Updated: 2024-12-13

Study Results

Results available

Outcome measurements, participant flow, baseline characteristics, and adverse events have been published for this study.

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE3

Total Enrollment

168 participants

Study Classification

INTERVENTIONAL

Study Start Date

2019-12-11

Study Completion Date

2023-07-12

Brief Summary

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The main objective of this study was to evaluate the efficacy of eplontersen as compared with the historical control of the placebo cohort in the NEURO-TTR trial (NCT01737398/2012-001831-30), in subjects with hereditary transthyretin-mediated amyloidosis polyneuropathy (hATTR-PN). For more information, please visit http://www.neuro-ttransform.com/.

Detailed Description

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This study was a multicenter, open-label study in up to 168 participants, who were randomized to receive subcutaneous (SC) injections of either eplontersen once every 4 weeks or inotersen once a week. Participants also received daily supplemental doses of the recommended daily allowance of vitamin A. Participants included in the inotersen reference arm crossed over to eplontersen at Week 37 after completing the Week 35 assessments.

Conditions

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Hereditary Transthyretin-Mediated Amyloid Polyneuropathy

Keywords

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Polyneuropathy Amyloidosis ATTR TTR

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

CROSSOVER

Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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Inotersen

Participants received inotersen, 300 milligrams (mg), subcutaneously (SC), once weekly up to Week 34. After Week 35 assessment, participants received eplontersen, 45 mg, SC, once every 4 weeks starting from Week 37 up to Week 81.

Group Type ACTIVE_COMPARATOR

Inotersen

Intervention Type DRUG

Inotersen by subcutaneous injection

Eplontersen

Intervention Type DRUG

Eplontersen by subcutaneous injection

Eplontersen

Participants received eplontersen, 45 mg, SC, once every 4 weeks up to Week 81.

Group Type EXPERIMENTAL

Eplontersen

Intervention Type DRUG

Eplontersen by subcutaneous injection

Interventions

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Inotersen

Inotersen by subcutaneous injection

Intervention Type DRUG

Eplontersen

Eplontersen by subcutaneous injection

Intervention Type DRUG

Other Intervention Names

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TEGSEDI ISIS 420915 ION-682884 AKCEA-TTR-LRx IONIS-TTR-LRx

Eligibility Criteria

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Inclusion Criteria

1. Aged 18 to 82 years at the time of informed consent
2. Females must be non-pregnant and non-lactating, and either surgically sterile or post-menopausal or abstinent
3. Males must be surgically sterile or, abstinent or, if engaged in sexual relations with a woman of child-bearing potential, the participant or the participantss non-pregnant female partner must be using a highly effective contraceptive method
4. Diagnosis of hereditary transthyretin-mediated polyneuropathy as defined by meeting all 3 of the following:

* Stage 1 or Stage 2 Familial Amyloid Polyneuropathy (FAP) or Coutinho Stage
* Documented genetic mutation in the TTR gene
* Symptoms and signs consistent with neuropathy associated with transthyretin amyloidosis, including Neuropathy Impairment Score (NIS) ≥ 10 and ≤ 130

Exclusion Criteria

1. Clinically-significant (CS) abnormalities in medical history, screening laboratory results, physical or physical examination that would render a participants unsuitable for inclusion, including but not limited to abnormal safety labs
2. Karnofsky performance status ≤ 50
3. Other causes of sensorimotor or autonomic neuropathy (e.g., autoimmune disease), including uncontrolled diabetes
4. Prior liver transplant or anticipated liver transplant within 1-yr of Screening
5. New York Heart Association (NYHA) functional classification of ≥ 3
6. Acute coronary syndrome within 6 months of screening or major surgery within 3 months of Screening
7. Other types of amyloidosis
8. Have any other conditions, which, in the opinion of the Investigator or Sponsor would make the participant unsuitable for inclusion, or could interfere with the participant participating in or completing the Study
9. Current treatment with any approved drug for hereditary TTR amyloidosis such as Vyndaqel® / Vyndamax™ (tafamidis), Tegsedi™ (inotersen), Onpattro™ (patisiran), off-label use of diflunisal or doxycycline, and tauroursodeoxycholic acid (TUDCA). If previously treated with Vyndaqel® / Vyndamax™, diflunisal or doxycycline, and TUDCA, must have discontinued treatment for at least 2 weeks prior to Study Day 1
10. Previous treatment with Tegsedi™ (Inotersen) or Onpattro™ (patisiran), or other oligonucleotide or RNA therapeutic (including siRNA)
Minimum Eligible Age

18 Years

Maximum Eligible Age

82 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Ionis Pharmaceuticals, Inc.

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Locations

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Mayo Clinic - Arizona

Scottsdale, Arizona, United States

Site Status

Mayo Clinic - Jacksonville

Jacksonville, Florida, United States

Site Status

Indiana University School of Medicine - Indianapolis

Indianapolis, Indiana, United States

Site Status

University of Kansas Medical Center

Kansas City, Kansas, United States

Site Status

Johns Hopkins University Neurology Research Office

Baltimore, Maryland, United States

Site Status

Boston University School of Medicine

Boston, Massachusetts, United States

Site Status

Mayo Clinic - Rochester

Rochester, Minnesota, United States

Site Status

The Neurological Institute of New York

New York, New York, United States

Site Status

University of North Carolina Hospitals - Neurology Clinic

Chapel Hill, North Carolina, United States

Site Status

Oregon Health and Science University

Portland, Oregon, United States

Site Status

Penn Presbyterian Medical Center

Philadelphia, Pennsylvania, United States

Site Status

University of Washington Medical Center

Seattle, Washington, United States

Site Status

Hospital Italiano de Buenos Aires

Ciudad Autónoma de Buenos Aires, Buenos Aires, Argentina

Site Status

Hospital El Cruce

San Juan Bautista, Buenos Aires, Argentina

Site Status

STAT Research

Buenos Aires, , Argentina

Site Status

Instituto Fleni

Buenos Aires, , Argentina

Site Status

Perron Institute for Neurological and Translational Science

Nedlands, Western Australia, Australia

Site Status

Instituto de Neurologia de Curitiba

Curitiba, Paraná, Brazil

Site Status

Universidade Estadual de Campinas

Campinas, , Brazil

Site Status

Hospital das Clínicas da Faculdade de Medicina de Ribeirão Preto

Ribeirão Preto, , Brazil

Site Status

Hospital Universitário Clementino Fraga Filho

Rio de Janeiro, , Brazil

Site Status

Associação de Assistência à Criança Deficiente - Unidade Lar Escola

São Paulo, , Brazil

Site Status

Toronto General Hospital

Toronto, Ontario, Canada

Site Status

The Cyprus Institute of Neurology and Genetics

Égkomi, , Cyprus

Site Status

Centre Hospitalier Universitaire de Toulouse

Toulouse, Haute-Garonne, France

Site Status

Hôpital de la Timone

Marseille, , France

Site Status

Hôpital Bicêtre

Le Kremlin-Bicêtre, Île-de-France Region, France

Site Status

Universitätsklinikum Würzburg

Würzburg, Bavaria, Germany

Site Status

Universitätsklinikum Heidelberg

Heidelberg, , Germany

Site Status

University General Hospital of Heraklion (PAGNI)

Heraklion, Crete, Greece

Site Status

Azienda Ospedaliera Universitaria Policlinico Gaetano Martino

Messina, , Italy

Site Status

Fondazione IRCCS Istituto Neurologico Carlo Besta

Milan, , Italy

Site Status

Fondazione IRCCS Policlinico San Matteo

Pavia, , Italy

Site Status

Fondazione Policlinico Universitario Agostino Gemelli

Roma, , Italy

Site Status

Auckland City Hospital

Grafton, Auckland, New Zealand

Site Status

Centro Hospitalar Universitário Lisboa Norte - Hospital De Santa Maria

Lisbon, , Portugal

Site Status

Centro Hospitalar Universitário do Porto - Hospital Geral de Santo Antonio

Porto, , Portugal

Site Status

Hospital Son Llàtzer

Palma de Mallorca, Balearic Islands, Spain

Site Status

Hospital Clínico San Carlos

Madrid, , Spain

Site Status

Norrlands Universitetssjukhus

Umeå, , Sweden

Site Status

Chang Gung Memorial Hospital - Linkou Branch

Taoyuan, Guishan District, Taiwan

Site Status

China Medical University Hospital

Taichung, Taichung, Taiwan

Site Status

Taipei Veterans General Hospital

Taipei City, Taipei, Taiwan

Site Status

National Taiwan University Hospital

Taipei, , Taiwan

Site Status

İstanbul Üniversitesi - Istanbul Tıp Fakültesi

Istanbul, , Turkey (Türkiye)

Site Status

Countries

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Japan United States Argentina Australia Brazil Canada Cyprus France Germany Greece Italy New Zealand Portugal Spain Sweden Taiwan Turkey (Türkiye)

References

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Coelho T, Marques W Jr, Dasgupta NR, Chao CC, Parman Y, Franca MC Jr, Guo YC, Wixner J, Ro LS, Calandra CR, Kowacs PA, Berk JL, Obici L, Barroso FA, Weiler M, Conceicao I, Jung SW, Buchele G, Brambatti M, Chen J, Hughes SG, Schneider E, Viney NJ, Masri A, Gertz MR, Ando Y, Gillmore JD, Khella S, Dyck PJB, Waddington Cruz M; NEURO-TTRansform Investigators. Eplontersen for Hereditary Transthyretin Amyloidosis With Polyneuropathy. JAMA. 2023 Oct 17;330(15):1448-1458. doi: 10.1001/jama.2023.18688.

Reference Type RESULT
PMID: 37768671 (View on PubMed)

Coelho T, Waddington Cruz M, Chao CC, Parman Y, Wixner J, Weiler M, Barroso FA, Dasgupta NR, Jung SW, Schneider E, Viney NJ, Dyck PJB, Ando Y, Gillmore JD, Khella S, Gertz MA, Obici L, Berk JL. Characteristics of Patients with Hereditary Transthyretin Amyloidosis-Polyneuropathy (ATTRv-PN) in NEURO-TTRansform, an Open-label Phase 3 Study of Eplontersen. Neurol Ther. 2023 Feb;12(1):267-287. doi: 10.1007/s40120-022-00414-z. Epub 2022 Dec 16.

Reference Type DERIVED
PMID: 36525140 (View on PubMed)

Coelho T, Ando Y, Benson MD, Berk JL, Waddington-Cruz M, Dyck PJ, Gillmore JD, Khella SL, Litchy WJ, Obici L, Monteiro C, Tai LJ, Viney NJ, Buchele G, Brambatti M, Jung SW, St L O'Dea L, Tsimikas S, Schneider E, Geary RS, Monia BP, Gertz M. Design and Rationale of the Global Phase 3 NEURO-TTRansform Study of Antisense Oligonucleotide AKCEA-TTR-LRx (ION-682884-CS3) in Hereditary Transthyretin-Mediated Amyloid Polyneuropathy. Neurol Ther. 2021 Jun;10(1):375-389. doi: 10.1007/s40120-021-00235-6. Epub 2021 Feb 26.

Reference Type DERIVED
PMID: 33638113 (View on PubMed)

Provided Documents

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Document Type: Study Protocol and Statistical Analysis Plan

View Document

Other Identifiers

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2019-001698-10

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

ION-682884-CS3

Identifier Type: -

Identifier Source: org_study_id