RECRUITING

The BENeFiTS Trial in Beta Thalassemia Intermedia

Description

Beta-thalassemias and hemoglobinopathies are serious inherited blood diseases caused by abnormal or deficiency of beta A chains of hemoglobin, the protein in red blood cells which delivers oxygen throughout the body.The diseases are characterized by hemolytic anemia, organ damage, and early mortality without treatment. Increases in another type of (normal) hemoglobin, fetal globin (HbF), which is normally silenced in infancy, reduces anemia and morbidity. Even incremental augmentation of fetal globin is established to reduce red blood cell pathology, anemia, certain complications, and to improve survival. This trial will evaluate an oral drug discovered in a high throughput screen, which increases fetal globin protein (HbF and red blood cells expressing HbF)and messenger ribonucleic acid (mRNA) to high levels in anemic nonhuman primates and in transgenic mice. The study drug acts by suppressing 4 repressors of the fetal globin gene promoter in progenitor cells from patients. The drug has been used for 50 years in a combination product for different actions - to enhance half-life and reduce side effects of a different active drug- and is considered safe for long-term use. This trial will first evaluate 3 dose levels in small cohorts of nontransfused patients with beta thalassemia intermedia. The most active dose will then be evaluated in larger subject groups with beta thalassemia and other hemoglobinopathies, such as sickle cell disease.

Study Overview

Study Details

Study overview

Beta-thalassemias and hemoglobinopathies are serious inherited blood diseases caused by abnormal or deficiency of beta A chains of hemoglobin, the protein in red blood cells which delivers oxygen throughout the body.The diseases are characterized by hemolytic anemia, organ damage, and early mortality without treatment. Increases in another type of (normal) hemoglobin, fetal globin (HbF), which is normally silenced in infancy, reduces anemia and morbidity. Even incremental augmentation of fetal globin is established to reduce red blood cell pathology, anemia, certain complications, and to improve survival. This trial will evaluate an oral drug discovered in a high throughput screen, which increases fetal globin protein (HbF and red blood cells expressing HbF)and messenger ribonucleic acid (mRNA) to high levels in anemic nonhuman primates and in transgenic mice. The study drug acts by suppressing 4 repressors of the fetal globin gene promoter in progenitor cells from patients. The drug has been used for 50 years in a combination product for different actions - to enhance half-life and reduce side effects of a different active drug- and is considered safe for long-term use. This trial will first evaluate 3 dose levels in small cohorts of nontransfused patients with beta thalassemia intermedia. The most active dose will then be evaluated in larger subject groups with beta thalassemia and other hemoglobinopathies, such as sickle cell disease.

A Phase 1b Sequential Open Label Dose-Ranging Study of Safety, Pharmacokinetics, and Preliminary Activity of Benserazide in Subjects With Beta Thalassemia Intermedia and Sickle Cell Disease

The BENeFiTS Trial in Beta Thalassemia Intermedia

Condition
Beta Thalassemia Intermedia
Intervention / Treatment

-

Contacts and Locations

Oakland

UCSF Benioff Children's Hospital at Oakland, Oakland, California, United States, 94609

Boston

Massachusetts General Hospital, Boston, Massachusetts, United States, 02114

Weston

Susan Perrine, Weston, Massachusetts, United States, 02493

New York

Weil Cornell Medicine, New York, New York, United States, 10065

Participation Criteria

Researchers look for people who fit a certain description, called eligibility criteria. Some examples of these criteria are a person's general health condition or prior treatments.

For general information about clinical research, read Learn About Studies.

Eligibility Criteria

  • * Beta thalassemia intermedia (BTI) or (NTDT, Non-Transfusion Dependent Thalassemia) with at least one documented beta thalassemia mutation, including HbE beta thalassemia or an established diagnosis of sickle cell disease
  • * \>18 years of age at time of consent
  • * Average of 2 total hemoglobin (Hgb) levels between 6.0 and 10.0 g/dL in the preceding 6 months
  • * Able and willing to give consent and comply with all study procedures
  • * If female and of childbearing potential, must have a documented negative pregnancy test prior to entry and agree to use one or more locally medically accepted methods of contraception
  • * Red blood cell (RBC) transfusion within 2 months prior to administration of study medication
  • * Participating in a chronic transfusion program
  • * Pulmonary hypertension requiring oxygen therapy
  • * Use of erythropoiesis stimulating agents within 90 days of first dose
  • * Transaminases \> 3 times upper limit of institution normal (ULN)
  • * Total and direct bilirubin \> 3 times institution ULN unless due solely to hemolysis
  • * Known infection with HIV or hepatitis C (untreated)
  • * Fever \> 38.5°C in the week prior to first administration of study medication
  • * History of osteoporosis or osteomalacia with a fragility fracture
  • * Received other investigational systemic therapy within 30 days prior to first dose
  • * Narrow angle glaucoma
  • * Currently pregnant or breast feeding a child
  • * Known current drug or alcohol abuse
  • * Taking monoamine oxidase inhibitors
  • * Other co-morbidity that substantially increases subject risk for the study per Investigator discretion

Ages Eligible for Study

18 Years to

Sexes Eligible for Study

ALL

Accepts Healthy Volunteers

No

Collaborators and Investigators

Phoenicia BioScience,

Susan Perrine, MD, STUDY_DIRECTOR, Phoenicia BioScience

Kevin Kuo, MD, PRINCIPAL_INVESTIGATOR, University Health Network, Toronto General Hospital

Sylvia Singer, MD, PRINCIPAL_INVESTIGATOR, UCSF Benioff Children's Hospital at Oakland

Hanny D Al-Samkari, MD, PRINCIPAL_INVESTIGATOR, Massachusetts General Hospital

Sujit Sheth, MD MS, PRINCIPAL_INVESTIGATOR, Weill Medical College of Cornell University

Study Record Dates

2025-02-28