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Phase 3 N=80 Treatment

A Study of Fitusiran in Severe Hemophilia A and B Patients Previously Receiving Factor or Bypassing Agent Prophylaxis

Hemophilia

Enrolled (actual)
80
Serious AEs
11.2%
Results posted
Feb 2023
Primary outcome: Primary: Estimated Annualized Bleeding Rate (ABR) — 7.482; 2.908 episodes per participant per year — p==0.0008

Study Design & Population

Study type
Interventional
Phase
Phase 3
Interventions
Fitusiran (Drug); BPA prophylaxis (Drug); Factor (FVIII or FIX) prophylaxis (Drug)
Age
Pediatric, Adult, Older Adult · 12+ yrs
Sex
Male
Sponsor
Genzyme, a Sanofi Company
Primary completion
Jan 2022

Outcome Measures

OutcomeResultp-value
PRIMARY
Estimated Annualized Bleeding Rate (ABR)
7.482; 2.908 =0.0008 sig
PRIMARY
Observed Annualized Bleeding Rate (ABR)
7.56; 3.19
SECONDARY
Estimated Annualized Spontaneous Bleeding Rate
5.002; 2.222
SECONDARY
Observed Annualized Spontaneous Bleeding Rate
5.09; 2.51
SECONDARY
Estimated Annualized Joint Bleeding Rate
5.282; 2.564
SECONDARY
Observed Annualized Joint Bleeding Rate
5.35; 2.82
SECONDARY
Change in Haemophilia Quality of Life Questionnaire for Adults (Haem-A-QOL) Physical Health Score in the Fitusiran Treatment Period and the Factor or BPA Prophylaxis Period
-6.00; -9.60
SECONDARY
Change in Haemophilia Quality of Life Questionnaire for Adults Total Score in the Fitusiran Treatment Period and the Factor or BPA Prophylaxis Period
-3.07; -7.62
SECONDARY
Estimated Annualized Bleeding Rate in the Fitusiran Onset Period
5.419
SECONDARY
Observed Annualized Bleeding Rate in the Fitusiran Onset Period
5.42
SECONDARY
Estimated Annualized Bleeding Rate in the Fitusiran Treatment Period
3.317
SECONDARY
Observed Annualized Bleeding Rate in the Fitusiran Treatment Period
3.48
SECONDARY
Cohort A: Annualized Weight-adjusted Consumption of BPA (Activated Prothrombin Complex Concentrates)
7912.7; 39.7
SECONDARY
Cohort A: Annualized Weight-adjusted Consumption of BPA (Recombinant Factor VIIa)
18895.8; 168.8
SECONDARY
Cohort B: Annualized Weight-adjusted Consumption of Factor
3396.9; 60.7; 3175.5; 17.8

Summary

Primary Objective: To characterize the frequency of bleeding episodes (BE) while receiving fitusiran treatment, relative to the frequency of bleeding episodes while receiving factor concentrate or bypassing agent (BPA) prophylaxis. Secondary Objectives: * To characterize the following while receiving fitusiran treatment, relative to receiving factor or BPA prophylaxis: * the frequency of spontaneous bleeding episodes * the frequency of joint bleeding episodes * health related quality of life (HRQOL) in participants greater than or equal to (>=) 17 years of age * To characterize the frequency of bleeding episodes during the onset and treatment periods in participants receiving fitusiran. * To characterize the safety and tolerability of fitusiran. * To characterize the annualized weight-adjusted consumption of factor/BPA while receiving fitusiran treatment, relative to receiving factor or BPA prophylaxis.

Eligibility Criteria

Inclusion Criteria

  • Males, >=12 years of age.
  • Severe hemophilia A or B (as evidenced by a central laboratory measurement at screening or documented medical record evidence of FVIII less than ( =0.6 Bethesda Unit per milliliter (BU/mL) at screening, or
  • Inhibitor titer of =0.6 BU/mL, or
  • Inhibitor titer of =20) prior to enrollment
  • In the opinion of the Investigator, with approval of Sponsor Medical Monitor, 6-month BPA prophylaxis period should be omitted.
  • Non-inhibitor: Use of factor concentrates for prophylaxis and for any bleeding episodes for at least the last 6 months prior to screening, and met each of the following criterion:
  • Nijmegen-modified Bethesda assay inhibitor titer of <0.6 BU/mL at screening and
  • No use of BPAs to treat bleeding episodes for at least the last 6 months prior to screening and
  • No history of immune tolerance induction therapy within the past 3 years prior to screening.
  • Documented prophylactic treatment with factor concentrates or BPAs for the treatment of hemophilia A or B for at least 6 months prior to screening.
  • Adherent to the prescribed prophylactic therapy for at least 6 months prior to screening per Investigator assessment.
  • Willed and complied with the study requirements and to provide written informed consent and assent.

Exclusion Criteria

  • Known co-existing bleeding disorders other than hemophilia A or B.
  • AT activity <60% at screening.
  • Co-existing thrombophilic disorder.
  • Clinically significant liver disease.
  • Active Hepatitis C virus infection.
  • Acute or chronic Hepatitis B virus infection.
  • HIV positive with a CD4 count of <200 cells per microliter.
  • History of arterial or venous thromboembolism.
  • Inadequate renal function.
  • History of multiple drug allergies or history of allergic reaction to an oligonucleotide or N-Acetylgalactosamine (GalNAc).
  • History of intolerance to subcutaneous injection(s).
  • Any other conditions or comorbidities that made the participant unsuitable for enrollment or could interfere with participation in or completion of the study, per Investigator judgment.
View full record on ClinicalTrials.gov →

Data sourced from ClinicalTrials.gov (NCT03549871). Outcome figures and adverse-event rates are extracted automatically from the registry's posted results and are provided for clinician reference, not as a substitute for the primary publication.

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