NCT05031780

Brief Summary

This clinical trial is a Phase 2/3 study that will determine the recommended dose of mitapivat and evaluate the efficacy and safety of mitapivat in sickle cell disease by testing how well mitapivat works compared to placebo to increase the amount of hemoglobin in the blood and to reduce or prevent the occurrence of sickle cell pain crises. In addition, the long-term effect of mitapivat on efficacy and safety will be explored in an open-label extension portion.

Trial Health

87
On Track

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
286

participants targeted

Target at P75+ for phase_2

Timeline
44mo left

Started Feb 2022

Longer than P75 for phase_2

Geographic Reach
16 countries

91 active sites

Status
active not recruiting

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

Study Progress54%
Feb 2022Feb 2030

First Submitted

Initial submission to the registry

July 29, 2021

Completed
1 month until next milestone

First Posted

Study publicly available on registry

September 2, 2021

Completed
5 months until next milestone

Study Start

First participant enrolled

February 11, 2022

Completed
3.7 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

October 30, 2025

Completed
4.3 years until next milestone

Study Completion

Last participant's last visit for all outcomes

February 1, 2030

Expected
Last Updated

May 29, 2026

Status Verified

May 1, 2026

Enrollment Period

3.7 years

First QC Date

July 29, 2021

Last Update Submit

May 28, 2026

Conditions

Outcome Measures

Primary Outcomes (4)

  • Phase 2: Percentage of Participants With Hemoglobin (Hb) Response

    Week 12

  • Phase 2: Percentage of Participants With Treatment-Emergent Adverse Events (AEs) and Treatment-Emergent Serious AEs (SAEs)

    Up to Week 12

  • Phase 3: Percentage of Participants With Hb Response

    Week 52

  • Phase 3: Annualized Rate of Sickle Cell Pain Crises (SCPCs)

    Up to Week 52

Secondary Outcomes (36)

  • Phase 2: Change From Baseline in Hb Concentration

    Baseline, Week 10 up to Week 12

  • Phase 2: Change From Baseline in Indirect Bilirubin

    Baseline, Week 10 up to Week 12

  • Phase 2: Change From Baseline in Lactate Dehydrogenase (LDH)

    Baseline, Week 10 up to Week 12

  • Phase 2: Change From Baseline in Absolute Reticulocytes Count

    Baseline, Week 10 up to Week 12

  • Phase 2: Change From Baseline in Percent Reticulocytes

    Baseline, Week 10 up to Week 12

  • +31 more secondary outcomes

Study Arms (7)

Phase 2: Mitapivat 50 mg BID

EXPERIMENTAL

Double-blind Period: Mitapivat 50 milligrams (mg) twice daily (BID) for 12 weeks.

Drug: Mitapivat

Phase 2: Mitapivat 100 mg BID

EXPERIMENTAL

Double-blind Period: Mitapivat 100 mg BID for 12 weeks.

Drug: Mitapivat

Phase 2: Placebo

PLACEBO COMPARATOR

Double-blind Period: Mitapivat-matching placebo for 12 weeks.

Other: Mitapivat-matching placebo

Phase 2: Open-Label Extension Period

EXPERIMENTAL

Participants who received mitapivat 50mg BID in the double-blind period may choose to receive mitapivat 50mg BID for 216 weeks after. Participants who received mitapivat 100mg BID in the double-blind period may choose to receive mitapivat 100 mg BID for 216 weeks after. Participants who received mitapivat-matching placebo in the double-blind period, may be randomized to receive either mitapivat 50 mg or 100 mg BID for 216 weeks after.

Drug: Mitapivat

Phase 3: Mitapivat 100 mg BID

EXPERIMENTAL

Double-blind Period: Mitapivat 100 mg BID for 52 weeks.

Drug: Mitapivat

Phase 3: Placebo

PLACEBO COMPARATOR

Double-blind Period: Mitapivat-matching placebo for 52 weeks.

Other: Mitapivat-matching placebo

Phase 3: Open-Label Extension Period

EXPERIMENTAL

Participants may choose to receive mitapivat 100 mg BID for 216 weeks after the Double-blind Period. Participants who received mitapivat-matching placebo in the double-blind period, may choose to receive mitapivat 100 mg BID for 216 weeks after the Double-blind Period.

Drug: Mitapivat

Interventions

Mitapivat tablets

Also known as: AG-348, Mitapivat Sulfate
Phase 2: Mitapivat 100 mg BIDPhase 2: Mitapivat 50 mg BIDPhase 2: Open-Label Extension PeriodPhase 3: Mitapivat 100 mg BIDPhase 3: Open-Label Extension Period

Placebo to match 50 mg or 100 mg tablets

Phase 2: Placebo

Eligibility Criteria

Age16 Years+
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)

You may qualify if:

  • Age 16 years or older (18 years or older \[France and Germany\]); participants age 16 or 17 years must physically have completed puberty;
  • Documented diagnosis of sickle cell disease (SCD) (HbSS, HbSC \[combined heterozygosity for hemoglobins S and C\], HbS/beta 0- thalassemia, HbS/ beta plus thalassemia, or other sickle cell syndrome variants);
  • At least 2 SCPCs and no more than 10 SCPCs in the past 12 months;
  • Hemoglobin at least 5.5 and 10.5 gram per deciliter (g/dL) at the most. Hemoglobin concentration must be based on an average of at least 2 Hb concentration measurements (separated by ≥7 days) collected during the Screening Period;
  • If taking hydroxyurea, the hydroxyurea dose must be stable for at least 90 days before starting study drug. Discontinuation of hydroxyurea requires a 90-day washout prior to informed assent/consent;
  • Women capable of becoming pregnant must agree to use 2 forms of contraception.

You may not qualify if:

  • Pregnant, breastfeeding, or parturient;
  • Receiving regularly scheduled transfusions;
  • Hepatobiliary disorders including but not limited to significant liver disease or gallbladder disease;
  • Severe kidney disease;
  • Prior exposure to gene therapy or prior bone marrow or stem cell transplantation;
  • Currently receiving treatment with a disease-modifying therapy for SCD (eg, voxelotor, crizanlizumab, L-glutamine), with the exception of hydroxyurea. The last dose of voxelotor, crizanlizumab, and L-glutamine must have been administered at least 90 days before randomization;
  • Currently receiving treatment with hematopoietic stimulating agents; the last dose must have been administered at least 90 days before starting study drug;
  • Received treatment on another investigational trial within 90 days prior to start of study drug or plans to participate in another investigational drug trial;
  • Taking medications that are strong inhibitors of CYP3A4/5 or strong inducers of CYP3A4 that cannot be stopped in an acceptable timeframe before starting study drug (timeframe will be discussed with your doctor).

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (91)

University of California San Diego

La Jolla, California, 92037-1337, United States

Location

UCLA Health

Los Angeles, California, 90095-1678, United States

Location

Children's Hospital Oakland

Oakland, California, 94609, United States

Location

University of Connecticut Health Center

Farmington, Connecticut, 06030-0001, United States

Location

Children's National Hospital

Washington D.C., District of Columbia, 20010-2916, United States

Location

MedStar Washington Hospital Center

Washington D.C., District of Columbia, 20010, United States

Location

Sylvester Comprehensive Cancer Center-Miami

Miami, Florida, 33101, United States

Location

University of Chicago Medical Center

Chicago, Illinois, 60637-1443, United States

Location

Riley Hospital For Children

Indianapolis, Indiana, 46202-5109, United States

Location

LSU Health Sciences Center - Shreveport

Shreveport, Louisiana, 71103-4228, United States

Location

National Heart Lung and Blood Institute

Bethesda, Maryland, 20814, United States

Location

Kaiser Permanente - Largo Medical Center

Largo, Maryland, 20774-5374, United States

Location

Massachusetts General Hospital

Boston, Massachusetts, 02114-2621, United States

Location

Boston Children's Hospital

Boston, Massachusetts, 02115-5724, United States

Location

Boston Medical Center & Boston University School of Medicine

Boston, Massachusetts, 02118, United States

Location

University of Michigan

Ann Arbor, Michigan, 48109-5000, United States

Location

Children's Hospital of Michigan

Detroit, Michigan, 48201, United States

Location

Southern Specialty Clinic

Flowood, Mississippi, 39232, United States

Location

Mississippi Center for Advanced Medicine

Madison, Mississippi, 39110-6115, United States

Location

Cure 4 The Kids Foundation, A Division of Roseman University of Health Sciences

Las Vegas, Nevada, 89106, United States

Location

East Carolina University - Brody School of Medicine

Greenville, North Carolina, 27834, United States

Location

Penn Medicine - University of Pennsylvania Health System

Philadelphia, Pennsylvania, 19104-5127, United States

Location

St. Christopher's Hospital for Children

Philadelphia, Pennsylvania, 19134-1011, United States

Location

Lifespan at Rhode Island Hospital

Providence, Rhode Island, 02903, United States

Location

University of Texas Health Science Center of Houston

Houston, Texas, 77030-1501, United States

Location

Texas Children's Hospital

Houston, Texas, 77030, United States

Location

Virginia Commonwealth University

Richmond, Virginia, 23298-5058, United States

Location

Seattle Cancer Care Alliance, University of Washington

Seattle, Washington, 98195, United States

Location

Hôpital Erasme

Anderlecht, Brussels Capital, 1070, Belgium

Location

Universitair Ziekenhuis Antwerpen

Edegem, Brussels Capital, 2650, Belgium

Location

ZAS Cadix

Antwerp, 2030, Belgium

Location

CHR de la Citadelle

Liège, 4000, Belgium

Location

Clinique CHC MontLégia

Liège, 4000, Belgium

Location

Multihemo Servicos Medicos S/A

Recife, Pernambuco, 50070-460, Brazil

Location

Hospital de Clinicas de Porto Alegre (HCPA) - PPDS

Porto Alegre, Rio Grande do Sul, 90035-903, Brazil

Location

Hospital Sao Lucas Da Pontificia Universidade Catolica Do Rio Grande Do Sul (PUCRS)

Porto Alegre, Rio Grande do Sul, 90619-900, Brazil

Location

Hospital de Clínicas da Unicamp

Campinas, São Paulo, 13083-878, Brazil

Location

Hospital Das Clínicas da Faculdade de Medicina de Ribeirão Preto - USP

Ribeirão Preto, São Paulo, 14051-140, Brazil

Location

Praxis Pesquisa Medica

Santo André, São Paulo, Brazil

Location

HEMORIO Instituto Nacional de Hematologia

Rio de Janeiro, 20211-030, Brazil

Location

Hospital das Clínicas da Faculdade de Medicina da Universidad de São Paulo

São Paulo, 05403-010, Brazil

Location

McMaster University - St. Joseph's Healthcare Hamilton

Hamilton, Ontario, L8N 3Z5, Canada

Location

University Health Network

Toronto, Ontario, M5G2C4, Canada

Location

CHU Montreal

Montreal, Quebec, H2X 3E4, Canada

Location

McGill University Health Center

Montreal, Quebec, H4A 3J1, Canada

Location

Hopitaux de La Timone

Marseille, Bouches-du-Rhône, 13005, France

Location

Hôpital Pellegrin, CHU de Bordeaux

Bordeaux, Gironde, 33000, France

Location

CHU Guadeloupe

Pointe à Pitre, Guadeloupe, 97139, France

Location

Institut Universitaire du Cancer de Toulouse - Oncopole

Toulouse, Haute-Garonne, 31059, France

Location

CHU Hôpital Henri Mondor

Créteil, Val-de-Marne, 94000, France

Location

Hôpital Européen Georges Pompidou

Paris, Île-de-France Region, 75015, France

Location

Universitätsklinikum Essen

Essen, 45147, Germany

Location

Universitätsklinikum Regensburg

Regensburg, 93053, Germany

Location

HaEmek Medical Center

Afula, 18101, Israel

Location

Rambam Medical Center

Haifa, Ḥeifā, 31096, Israel

Location

Ziv Medical Center

Safed, Ḥeifā, 13100, Israel

Location

A.O.R.N. "A. Cardarelli"

Naples, Campania, 80131, Italy

Location

AOU dell'Università degli Studi della Campania Luigi Vanvitelli

Naples, Campania, 80138, Italy

Location

Azienda Ospedaliero Universitaria Di Modena Policlinico

Modena, Emilia-Romagna, 41100, Italy

Location

IRCCS Ospedale Pediatrico Bambino Gesù - INCIPIT - PIN

Rome, Lazio, 00165, Italy

Location

Ente Ospedaliero Ospedali Galliera

Genoa, Liguria, 16128, Italy

Location

Azienda Ospedaliera Ospedali Riuniti Villa Sofia-Cervello

Palermo, Sicily, 90146, Italy

Location

Kemri Usamru

Kisumu, Western, 40100, Kenya

Location

Kondele Children's Hospital

Kisumu, 40100, Kenya

Location

Victoria Biomedical Research Institute (VIBRI)

Kisumu, 40100, Kenya

Location

KEMRI CRDR Clinical Research Clinic Nairobi

Nairobi, 00100, Kenya

Location

KEMRI/CRDR Siaya Clinical Research Annex

Nairobi, 00200, Kenya

Location

Strathmore University

Nairobi, 00200, Kenya

Location

Gertrude's Children's Hospital

Nairobi, 42325- 00100, Kenya

Location

American University of Beirut Medical Center

Beirut, Beirut, 11-0236, Lebanon

Location

Nini Hospital

Tarablus, North Lebanon, 1434, Lebanon

Location

American University of Beirut Medical Center

Beirut, 4407-2020, Lebanon

Location

Hammoud Hospital University Medical Center

Sidon, H96G+247, Lebanon

Location

Erasmus MC

Rotterdam, South Holland, 3015 GD, Netherlands

Location

Universitair Medisch Centrum Utrecht

Utrecht, 3584 CX, Netherlands

Location

National Hospital Abuja

Abuja, Federal Capital Territory, 900271, Nigeria

Location

University of Abuja Teaching Hospital

Abuja, Federal Capital Territory, 900271, Nigeria

Location

Lagos University Teaching Hospital

Surulere, Lagos, 101014, Nigeria

Location

Sultan Qaboos University Hospital, Hematology Department, COM&HS

Muscat, Musqal, H5QC+36M, Oman

Location

King Khalid University Hospital

Riyadh, Ar Riya, 11472, Saudi Arabia

Location

King Abdullah International Medical Research Center

Riyadh, 1515 (KAIMRC), Saudi Arabia

Location

Hacettepe Universitesi Tip Fakultesi Hastanesi

Ankara, Adana, 06200, Turkey (Türkiye)

Location

Hacettepe University

Ankara, Adana, Turkey (Türkiye)

Location

Acibadem Adana Hospital

Seyhan, Adana, 01130, Turkey (Türkiye)

Location

Evelina Children's Hospital

London, City of London, SE1 7EH, United Kingdom

Location

Cambridge University Hospitals NHS Foundation Trust

Cambridge, CB2 0QQ, United Kingdom

Location

Guy's and St Thomas' NHS Foundation Trust

London, SE1 7EH, United Kingdom

Location

King's College Hospital NHS Foundation Trust

London, SE5 9RS, United Kingdom

Location

Hammersmith Hospital

London, W12 0HS, United Kingdom

Location

University College London Hospitals (UCLH)

London, WC1E 6BT, United Kingdom

Location

Manchester Royal Infirmary, Manchester University NHS Foundation Trust

Manchester, M13 9WL, United Kingdom

Location

Related Publications (4)

  • Conrey A, Asomaning N, Frey I, Charles RP, Lovins D, Xu JZ, Mendez-Marti S, Le K, Kruah B, Li Q, Dunkelberger E, Cellmer T, Yates A, Wind-Rotolo M, Huston C, Jeffries N, Eaton WA, Thein SL. Long-term mitapivat treatment is safe and efficacious in patients with sickle cell disease. Blood Red Cells Iron. 2025 Sep;1(2):100014. doi: 10.1016/j.brci.2025.100014. Epub 2025 Sep 11.

  • Idowu M, Otieno L, Dumitriu B, Lobo CLC, Thein SL, Andemariam B, Nnodu OE, Inati A, Glaros AK, Bartolucci P, Colombatti R, Taher AT, Abboud MR, Darbari D, Ataga KI, Antmen AB, Kuo KHM, de Souza Medina S, Oluyadi A, Iyer V, Morris S, Yates AM, Shao H, Patil S, Urbstonaitis R, Zaidi AU, Gheuens S, Smith WR. Safety and efficacy of mitapivat in sickle cell disease (RISE UP): results from the phase 2 portion of a global, double-blind, randomised, placebo-controlled trial. Lancet Haematol. 2025 Jan;12(1):e35-e44. doi: 10.1016/S2352-3026(24)00319-3. Epub 2024 Dec 4.

  • van Dijk MJ, Rab MAE, van Oirschot BA, Bos J, Derichs C, Rijneveld AW, Cnossen MH, Nur E, Biemond BJ, Bartels M, Jans JJM, van Solinge WW, Schutgens REG, van Wijk R, van Beers EJ. One-year safety and efficacy of mitapivat in sickle cell disease: follow-up results of a phase 2, open-label study. Blood Adv. 2023 Dec 26;7(24):7539-7550. doi: 10.1182/bloodadvances.2023011477.

  • Obadina M, Wilson S, Derebail VK, Little J. Emerging Therapies and Advances in Sickle Cell Disease with a Focus on Renal Manifestations. Kidney360. 2023 Jul 1;4(7):997-1005. doi: 10.34067/KID.0000000000000162. Epub 2023 May 31.

MeSH Terms

Conditions

Anemia, Sickle Cell

Interventions

mitapivat

Condition Hierarchy (Ancestors)

Anemia, Hemolytic, CongenitalAnemia, HemolyticAnemiaHematologic DiseasesHemic and Lymphatic DiseasesHemoglobinopathiesGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and Abnormalities

Study Design

Study Type
interventional
Phase
phase 2
Allocation
RANDOMIZED
Masking
QUADRUPLE
Who Masked
PARTICIPANT, CARE PROVIDER, INVESTIGATOR, OUTCOMES ASSESSOR
Purpose
TREATMENT
Intervention Model
SEQUENTIAL
Sponsor Type
INDUSTRY
Responsible Party
SPONSOR

Study Record Dates

First Submitted

July 29, 2021

First Posted

September 2, 2021

Study Start

February 11, 2022

Primary Completion

October 30, 2025

Study Completion (Estimated)

February 1, 2030

Last Updated

May 29, 2026

Record last verified: 2026-05

Data Sharing

IPD Sharing
Will not share

Locations