A Study to Determine the Efficacy and Safety of Luspatercept in Adults With Non Transfusion Dependent Beta (β)-Thalassemia

NCT ID: NCT03342404

Last Updated: 2023-12-20

Study Results

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE2

Total Enrollment

145 participants

Study Classification

INTERVENTIONAL

Study Start Date

2018-02-05

Study Completion Date

2022-11-28

Brief Summary

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This is a Phase 2, double-blind, randomized, placebo-controlled, multicenter study to determine the efficacy and safety of luspatercept (ACE-536) versus placebo in adults with non-transfusion dependent beta (β)-thalassemia. The study is divided into the Screening Period, Double-blind Treatment Period (DBTP), Open-label Phase (OLP), and Post-Treatment Follow-up Period (PTFP).

It is planned to randomize approximately 150 subjects at a 2:1 ratio of luspatercept versus placebo.

Detailed Description

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The primary objective is:

\- To evaluate the effect of luspatercept (BMS-986346) versus placebo on anemia, as measured by mean hemoglobin concentration in the absence of transfusions over a continuous 12-week interval, from Week 13 to Week 24, compared to baseline.

The secondary objectives are:

* To evaluate the effect of luspatercept versus placebo in anemia-related symptoms in participants with β-thalassemia, as measured by non-transfusion dependent β-thalassemia-patient reported outcome (NTDT-PRO) over continuous 12-week intervals (from Weeks 13 to 24 and from Weeks 37 to 48) compared to baseline.
* To evaluate the effect of luspatercept versus placebo on functional and health-related quality of life (QoL) as measured by Medical Outcomes Study 36-Item Short Form (SF-36) and Functional Assessment of Chronic Illness Therapy-Fatigue (FACIT-F) questionnaires
* To evaluate the long-term effect of luspatercept versus placebo on anemia, as measured by mean hemoglobin concentration in the absence of transfusions over a continuous 12-week interval from Week 37 to Week 48, compared to baseline
* To evaluate the effect of luspatercept versus placebo on iron overload, as measured by liver iron concentration (LIC) and iron chelation therapy (ICT) daily dose
* To evaluate the effect of luspatercept versus placebo on iron overload, as measured by serum ferritin
* To evaluate the duration of erythroid response
* To evaluate the effect of luspatercept versus placebo on physical activity measured by 6-minute walk test (6MWT)

Safety and Pharmacokinetics (PK) Objectives

* To evaluate safety and tolerability of luspatercept, including immunogenicity
* To evaluate population pharmacokinetics (PK) of luspatercept in subjects with β-thalassemia

The exploratory objectives are:

* To evaluate the effect of luspatercept versus placebo on measures of extra-medullary hematopoietic (EMH) masses, bone mineral density, splenomegaly, pulmonary hypertension, and leg ulcers, when present
* To evaluate the effect of luspatercept versus placebo on the β-thalassemia severity as measured by the NTDT severity score system
* To evaluate the effect of luspatercept versus placebo on the β-thalassemia severity as measured by the Morbidity-free Survival parameters
* To examine the relationship of baseline and change in serum Growth Differentiation Factor 11 (GDF11) and other related biomarkers with response to treatment with luspatercept
* To examine the effect of luspatercept on fetal hemoglobin (HbF)
* To examine the effect of luspatercept on Health Resource Utilization (HRU)

Conditions

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Thalassemia

Keywords

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(β)-Thalassemia Beta-Thalassemia Phase 2 Luspatercept ACE-536 Safety Efficacy Placebo Red Blood Cell Transfusions Erythrocyte transfusion Hb increase NTDT

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors

Study Groups

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Luspatercept (ACE-536) plus Best Supportive Care (BSC)

Arm Description: Luspatercept, subcutaneous(ly) (SC) once every 21 days

Group Type EXPERIMENTAL

Luspatercept

Intervention Type DRUG

Subjects will start with luspatercept at 1 mg/kg dose level every 3 weeks and can be dose escalated up to 1.25 mg/kg.

Best Supportive Care (BSC)

Intervention Type OTHER

Best Supportive Care (BSC)

Placebo plus Best Supportive Care (BSC)

normal saline solution subcutaneous(ly) (SC) once every 21 days

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type OTHER

Placebo, Subcutaneous, every 21 days

Best Supportive Care (BSC)

Intervention Type OTHER

Best Supportive Care (BSC)

Interventions

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Luspatercept

Subjects will start with luspatercept at 1 mg/kg dose level every 3 weeks and can be dose escalated up to 1.25 mg/kg.

Intervention Type DRUG

Placebo

Placebo, Subcutaneous, every 21 days

Intervention Type OTHER

Best Supportive Care (BSC)

Best Supportive Care (BSC)

Intervention Type OTHER

Other Intervention Names

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ACE-536 normal saline

Eligibility Criteria

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Inclusion Criteria

Subjects must satisfy the following criteria to be enrolled in the study:

1. Subjects must be ≥ 18 years of age at the time of signing the informed consent document (ICF).
2. Subject must understand and voluntarily sign an ICF prior to any study-related assessments/procedures being conducted.
3. Subject is willing and able to adhere to the study visit schedule (eg, not scheduled to receive hematopoietic stem cell transplantation) and other protocol requirements.
4. Subject must have documented diagnosis of β-thalassemia or hemoglobin E/ β-thalassemia. Concomitant alpha globin mutation and/or duplication are allowed.
5. Subject must be non-transfusion dependent, defined as 0 to 5 units of RBCs received during the 24-week period prior to randomization. Note: 1 unit defined for this entry criterion as approximately 200 to 350 mL of transfused packed RBCs.
6. Subject must not be on a regular transfusion program and must be RBC transfusion-free for at least ≥ 8 weeks prior to randomization
7. Subject must have mean baseline hemoglobin ≤ 10 g/dL, based on a minimum of 2 measurements ≥ 1 week apart within 4 weeks prior to randomization; hemoglobin values within 21 days post-transfusion will be excluded.
8. Subject must have performance status: Eastern Cooperative Oncology Group (ECOG) performance score of 0 to 1.
9. A female of childbearing potential (FCBP) for this study is defined as a female who:

1\) has achieved menarche at some point, 2) has not undergone a hysterectomy or bilateral oophorectomy or 3) has not been naturally postmenopausal (amenorrhea following cancer therapy does not rule out childbearing potential) for at least 24 consecutive months (ie, has had menses at any time in the preceding 24 consecutive months). A FCBP participating in the study must:

1. Have 2 negative pregnancy tests as verified by the Investigator prior to starting study therapy. She must agree to ongoing pregnancy testing during the course of the study, and after end of study treatment. This applies even if the subject practices true abstinence\* from heterosexual contact.
2. Either commit to true abstinence\* from heterosexual contact (which must be reviewed on a monthly basis and source documented). If a FCBP engages in sexual activity that may result in a pregnancy, she must agree to use, and be able to comply during the study therapy (including dose interruptions), and for 12 weeks (approximately 5 times the mean terminal half-life of luspatercept based on multiple dose pharmacokinetics \[PK\] data) after discontinuation of study therapy.

10\. Male subjects must:

a. Practice true abstinence (which must be reviewed on a monthly basis) or agree to use a condom during sexual contact with a pregnant female or a female of childbearing potential while participating in the study, during dose interruptions and for at least 12 weeks (approximately 5 times the mean terminal half-life of luspatercept based on multiple-dose PK data) following IP discontinuation, even if he has undergone a successful vasectomy

Exclusion Criteria

The presence of any of the following will exclude a subject from enrollment:

1. Any significant medical condition, laboratory abnormality, or psychiatric illness that would prevent the subject from participating in the study.
2. Any condition including the presence of laboratory abnormalities, which places the subject at unacceptable risk if he/she were to participate in the study.
3. Any condition that confounds the ability to interpret data from the study.
4. Diagnosis of hemoglobin S/β-thalassemia or alpha (α)-thalassemia (eg,Hemoglobin H).
5. Active hepatitis C (HCV) infection
6. Deep vein thrombosis (DVT) or stroke requiring medical intervention ≤ 24 weeks prior to randomization.
7. Subjects on chronic anticoagulant therapy are excluded, unless they stopped the treatment at least 28 days prior to randomization. Anticoagulant therapies for prophylaxis and for surgery or high-risk procedures as well as low molecular weight (LMW) heparin for superficial vein thrombosis (SVT) and chronic aspirin are allowed before and during the study.
8. Treatment with another investigational drug or device ≤ 28 days prior to randomization.
9. Prior exposure to sotatercept (ACE-011) or luspatercept (ACE-536).
10. Platelet count \> 1000 x 109/L.
11. Subjects on iron chelation therapy (ICT) at the time of ICF signature must have initiated the treatment with ICT at least 24 weeks before the predicted randomization date. ICT can be initiated at any time during treatment and should be used according to the label.
12. Subject had Hydroxyurea and ESA treatment ≤ 24 weeks prior to randomization, and no prior gene therapy.
13. Subject is pregnant or a lactating female.
14. Uncontrolled hypertension. Controlled hypertension for this protocol is considered ≤ Grade 1 according to National Cancer Institute Common Terminology for Adverse Events (NCI CTCAE) version 4.0 (current active minor version).
15. Subject has major organ damage, including:

1. Liver disease with alanine aminotransferase (ALT) \> 3 x upper limit of normal (ULN) or history/evidence of cirrhosis, as well as presence of masses/tumor detected by ultrasound at screening.
2. Heart disease, heart failure as classified by the New York Heart Association (NYHA) classification 3 or higher, or significant arrhythmia requiring treatment, or recent myocardial infarction (MI) within 6 months of randomization.
3. Severe lung disease, including pulmonary fibrosis or pulmonary hypertension, ie, ≥G3 NCI CTCAE version 4.0 (current active minor version).
4. Estimated glomerular filtration rate (eGFR) \< 60 ml/min/1.73 m2 (per Modification of Diet in Renal Disease \[MDRD\] formula).
16. Subject has received chronic systemic glucocorticoids ≤ 12 weeks prior to randomization (physiologic replacement therapy for adrenal insufficiency is allowed).
17. Major surgery ≤ 12 weeks prior to randomization (subjects must have completely recovered from any previous surgery prior to randomization).
18. History of severe allergic or anaphylactic reactions or hypersensitivity to recombinant proteins or excipients in the investigational product (see Investigator Brochure).
19. Subject has received immunosuppressants ≤ 28 days prior to randomization.
20. History or current malignancies (solid tumors and hematological malignancies) unless the subject has been free of the disease (including completion of any active or adjuvant treatment for prior malignancy) for ≥ 5 years. However, subjects with the following history/concurrent conditions are allowed:

* Basal or squamous cell carcinoma of the skin
* Carcinoma in situ of the cervix
* Carcinoma in situ of the breast
* Incidental histologic finding of prostate cancer (T1a or T1b using the tumor, nodes, metastasis \[TNM\] clinical staging system)
Minimum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Acceleron Pharma, Inc., a wholly-owned subsidiary of Merck & Co., Inc., Rahway, NJ USA

INDUSTRY

Sponsor Role collaborator

Celgene

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Jeevan Shetty, MD

Role: STUDY_DIRECTOR

Celgene Corporation

Locations

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Children's Hospital of Los Angeles

Los Angeles, California, United States

Site Status

Local Institution - 501

Los Angeles, California, United States

Site Status

Children's Hospital and Research Center at Oakland

Oakland, California, United States

Site Status

Local Institution - 503

Chicago, Illinois, United States

Site Status

Local Institution - 102

Athens, , Greece

Site Status

Local Institution - 101

Athens, , Greece

Site Status

Local Institution - 205

Cagliari, , Italy

Site Status

Universita degli Studi di Cagliari - ASL8

Cagliari, , Italy

Site Status

Local Institution - 202

Genoa, , Italy

Site Status

Local Institution - 201

Milan, , Italy

Site Status

Local Institution - 203

Naples, , Italy

Site Status

Local Institution - 206

Napoli, , Italy

Site Status

Local Institution - 204

Orbassano, , Italy

Site Status

Local Institution - 301

Hazmiyeh, , Lebanon

Site Status

Local Institution - 401

Bangkok, , Thailand

Site Status

Local Institution - 601

London Bloomsbury, , United Kingdom

Site Status

Countries

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United States Greece Italy Lebanon Thailand United Kingdom

References

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Piga A, Perrotta S, Gamberini MR, Voskaridou E, Melpignano A, Filosa A, Caruso V, Pietrangelo A, Longo F, Tartaglione I, Borgna-Pignatti C, Zhang X, Laadem A, Sherman ML, Attie KM. Luspatercept improves hemoglobin levels and blood transfusion requirements in a study of patients with beta-thalassemia. Blood. 2019 Mar 21;133(12):1279-1289. doi: 10.1182/blood-2018-10-879247. Epub 2019 Jan 7.

Reference Type BACKGROUND
PMID: 30617198 (View on PubMed)

Taher AT, Viprakasit V, Kattamis A, Perrotta S, Ricchi P, Porter JB, Coates TD, Forni GL, Musallam KM, Esposito O, Pilot R, Kuo WL, Lai Y, Reverte M, Wei R, Moro Bueno L, Cappellini MD. Efficacy and safety of luspatercept in non-transfusion-dependent beta-thalassemia: long-term results from the BEYOND study. Blood Adv. 2025 Aug 28:bloodadvances.2025016554. doi: 10.1182/bloodadvances.2025016554. Online ahead of print.

Reference Type DERIVED
PMID: 40875596 (View on PubMed)

Denton CC, Vodala S, Veluswamy S, Hofstra TC, Coates TD, Wood JC. Splenic iron decreases without change in volume or liver parameters during luspatercept therapy. Blood. 2023 Nov 30;142(22):1932-1934. doi: 10.1182/blood.2023021839.

Reference Type DERIVED
PMID: 37704579 (View on PubMed)

Taher AT, Musallam KM, Viprakasit V, Kattamis A, Lord-Bessen J, Yucel A, Guo S, Pelligra C, Shields AL, Shetty JK, Miteva D, Bueno LM, Cappellini MD. Psychometric evaluation of the NTDT-PRO questionnaire for assessing symptoms in patients with non-transfusion-dependent beta-thalassaemia. BMJ Open. 2023 Mar 22;13(3):e066683. doi: 10.1136/bmjopen-2022-066683.

Reference Type DERIVED
PMID: 36948565 (View on PubMed)

Taher AT, Cappellini MD, Kattamis A, Voskaridou E, Perrotta S, Piga AG, Filosa A, Porter JB, Coates TD, Forni GL, Thompson AA, Tartaglione I, Musallam KM, Backstrom JT, Esposito O, Giuseppi AC, Kuo WL, Miteva D, Lord-Bessen J, Yucel A, Zinger T, Shetty JK, Viprakasit V; BEYOND Investigators. Luspatercept for the treatment of anaemia in non-transfusion-dependent beta-thalassaemia (BEYOND): a phase 2, randomised, double-blind, multicentre, placebo-controlled trial. Lancet Haematol. 2022 Oct;9(10):e733-e744. doi: 10.1016/S2352-3026(22)00208-3. Epub 2022 Aug 22.

Reference Type DERIVED
PMID: 36007538 (View on PubMed)

Provided Documents

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Document Type: Study Protocol

View Document

Document Type: Statistical Analysis Plan

View Document

Related Links

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Other Identifiers

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U1111-1202-7068

Identifier Type: REGISTRY

Identifier Source: secondary_id

2015-003225-33

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

ACE-536-B-THAL-002

Identifier Type: -

Identifier Source: org_study_id