MedPath

BAX 326 Pediatric Study

Phase 2
Completed
Conditions
Hemophilia B
Interventions
Biological: BAX326
Registration Number
NCT01488994
Lead Sponsor
Baxalta now part of Shire
Brief Summary

The purpose of this study is to assess BAX 326 pharmacokinetic parameters, to evaluate its hemostatic efficacy, safety, immunogenicity, and changes in health-related quality of life in pediatric patients.

Detailed Description

The secondary outcome measure: Area Under the Plasma Concentration Versus Time Curve From 0 to 72 Hours (h) Post-infusion analysis was not done due to the different time-points for the last PK blood sample, AUC0-72 h was redundant and only total AUC was included in the PK analysis.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
23
Inclusion Criteria
  • Participant and/or legal representative has/have voluntarily provided signed informed consent
  • Participant has severe (FIX level < 1%) or moderately severe (FIX level ≤ 2%) hemophilia B
  • Participant is < 12 years old at the time of screening
  • Participant has no evidence of a history of FIX inhibitors (based on the participant's medical records)
  • Participant is immunocompetent as evidenced by a CD4 count ≥ 200 cells/mm^3

Main

Exclusion Criteria
  • Participant has a detectable FIX inhibitor at screening, with a titer ≥ 0.6 Bethesda Unit (BU)
  • Participant has a history of allergic reaction, e.g. anaphylaxis, following exposure to FIX concentrate(s)
  • Participant has evidence of an ongoing or recent thrombotic disease
  • Participant has an inherited or acquired hemostatic defect other than hemophilia B

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
BAX326 < 6 years of ageBAX326-
BAX326 6 to <12 years of ageBAX326-
Primary Outcome Measures
NameTimeMethod
Adverse Events (AEs) Possibly or Probably Related to BAX326Throughout study period (approximately 17 months)
Secondary Outcome Measures
NameTimeMethod
Pharmacokinetics (PK): Total Area Under the Plasma Concentration Versus Time Curve From 0 to Infinity Post-infusion Per Dose (Total AUC/Dose)Within 30 mins pre-infusion and 4 post-infusion timepoints. Refer to Population Description below for more details.
Pharmacokinetics (PK): Factor IX (FIX) Clearance (CL)Within 30 mins pre-infusion and 4 post-infusion timepoints. Refer to Population Description below for more details.

Computed as the dose divided by total Area under the curve (AUC)

Hemostatic Efficacy: Prophylaxis: Annualized Bleeding Rate (ABR)Throughout study period (approximately 17 months)

The annualized bleeding rate (ABR) during prophylaxis was calculated only for participants who had adequate treatment time for bleeding rate assessment (i.e., more than 3 months of prophylaxis treatment). The observation period for prophylaxis was to be the time between the first and the last prophylactic infusions. The treatment period for surgery was to be excluded from the bleed rate calculation. ABR calculated as (Number of bleeding episodes/observed treatment period in days) \* 365.25.

Pharmacokinetics (PK): Mean Residence Time (MRT)Within 30 mins pre-infusion and 4 post-infusion timepoints. Refer to Population Description below for more details.

Computed as total area under the first moment curve (total AUMC) divided by the total area under the concentration versus time curve (total AUC)

Pharmacokinetics (PK): Incremental Recovery (IR)Within 30 mins pre-infusion and 30 mins post-infusion

The rise in FIX activity in IU/dL per unit dose administered in IU/kg. Calculated as follows: (FIX activity at post-infusion minus FIX activity at pre-infusion) divided by weight-adjusted dose

Pharmacokinetics (PK): Elimination Phase Half-life (T 1/2)Within 30 mins pre-infusion and 4 post-infusion timepoints. Refer to Population Description below for more details.

Calculated as log_e2/λ, where λ is the regression slope in the terminal phase of the least absolute deviations regression model

Consumption of BAX326: Weight-adjusted Consumption Per Year (Annualized)Throughout study period (approximately 17 months)
Pharmacokinetics (PK): Incremental Recovery (IR) Over TimeWithin 30 mins pre-infusion and 30 mins post-infusion at baseline, Week 5, Week 13 and Week 26.

IR calculated as follows: (FIX activity at post-infusion minus FIX activity at pre-infusion) divided by weight-adjusted dose. IR is determined at baseline (PK analysis), Week 5, Week 13 and Week 26 timepoints. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \> 6 years of age; pediatric participants 6 to \<12 years of age; pharmacokinetic Full Analysis Set (PKFAS).

Safety: Number of Participants With Severe Allergic Reactions, e.g. AnaphylaxisThroughout study period (approximately 17 months)
Safety: Number of Participants With Thrombotic EventsThroughout study period (approximately 17 months)
Safety: Number of Participants Who Developed Antibodies to Chinese Hamster Ovary (CHO) Proteins and Recombinant Furin (rFurin)Throughout study period (approximately 17 months)

If more than 2-dilution increase as compared to pre-study level at screening and titers verified for specificity in the confirmatory assay.

Pharmacokinetics (PK): Total Area Under the Plasma Concentration Versus Time Curve From 0 to 72 Hours Post-infusion Per Dose (AUC 0-72h/Dose)Within 30 mins pre-infusion and 4 post-infusion timepoints
Hemostatic Efficacy: Treatment of Bleeding Episodes: Overall Hemostatic Efficacy Rating at Resolution of BleedThroughout study period (approximately 17 months)

Rating Scale for Treatment of bleeding episodes (4-point ordinal scale): - Excellent: Full relief of pain and cessation of objective signs of bleeding (eg, swelling, tenderness, and decreased range of motion in the case of musculoskeletal hemorrhage) after a single infusion. No additional infusion required for the control of bleeding. Administration of further infusions to maintain hemostasis did not affect this scoring. - Good: Definite pain relief and/or improvement in signs of bleeding after a single infusion. Possibly requires more than 1 infusion for complete resolution. - Fair: Probable and/or slight relief of pain and slight improvement in signs of bleeding after single infusion. Required more than 1 infusion for complete resolution. - None: No improvement or condition worsens.

Consumption of BAX326: Weight-adjusted Consumption Per MonthThroughout study period (approximately 17 months)
Safety and Immunogenicity: Number of Participants Who Developed Total Binding Antibodies to Factor IX (FIX)Throughout study period (approximately 17 months)

If more than 2-dilution increase as compared to pre-study level at screening and titers verified for specificity in the confirmatory assay. AB=antibodies in category for outcome measure data.

Health-related Quality of Life (HRQoL): Haemo-QoL, Change From Baseline in Total ScoreBaseline and 6 months

The Haemo-QoL is a quality of life (QoL) assessment instrument for children and adolescents with haemophilia. As a hemophilia-specific instrument, this measure assesses very specific aspects of dealing with hemophilia. For the Haemo-QoL, higher scores indicate a worse quality of life. Scores on a scale range between 0 and 100.

Health Resource Use: Emergency Room VisitsBaseline (Pharmacokinetic [PK] assessment), Week 5, Week 13 and Week 26

The number of Emergency Room visits per participant. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \< 6 years of age; pediatric participants 6 to \<12 years of age; Full Analysis Set.

Pharmacokinetics (PK): Volume of Distribution at Steady State (Vss)Within 30 mins pre-infusion and 4 post-infusion timepoints. Refer to Population Description below for more details.

Computed as Clearance (CL) \* Mean residence time (MRT)

Hemostatic Efficacy: Treatment of Bleeding Episodes: Number of Infusions Per Bleeding EpisodeThroughout study period (approximately 17 months)
Consumption of BAX326: Number of Infusions Per MonthThroughout study period (approximately 17 months)
Safety: Number of Participants With Clinically Significant Changes in Routine Laboratory Parameters (Haematology and Clinical Chemistry), and Vital SignsThroughout study period (approximately 17 months)

Categories consist of Clinically Significant (CS) changes in haemaotology parameters, clinical chemistry parameters and vital signs. Abbreviations in categories; Clin=clinical; params=parameters

Consumption of BAX326: Number of Infusions Per YearThroughout study period (approximately 17 months)
Consumption of BAX326: Weight-adjusted Consumption Per EventThroughout study period (approximately 17 months)

Event includes prophylactic infusions of study product and infusions of study product for treatment of bleeding episodes (BEs).

Safety and Immunogenicity: Number of Participants Who Developed Inhibitory Antibodies to Factor IX (FIX)Throughout study period (approximately 17 months)
Health Resource Use: Number of HospitalizationsBaseline (Pharmacokinetic [PK] assessment), Week 5, Week 13 and Week 26

The number of hospitalizations per participant. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \< 6 years of age; pediatric participants 6 to \<12 years of age; Full Analysis Set.

Health Resource Use: Length of HospitalizationBaseline (Pharmacokinetic [PK] assessment), Week 5, Week 13 and Week 26

The length of hospitalization per participant. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \< 6 years of age; pediatric participants 6 to \<12 years of age; Full Analysis Set.

Health-related Quality of Life (HRQoL): PedsQL™ Change From Baseline in Total ScoreBaseline and 6 months

For this study, the PedsQL™ questionnaires for participants 2 to 7 years of age (parent-proxy versions for age groups 2-4 years and 5-7 years) and PedsQL™ Child version for participants 8 to 12 years of age were used. The Peds-QL is a generic Health-Related Quality of Life (HR QoL) instrument designed specifically for a pediatric population. It captures the following domains: general health/activities, feelings/emotional, social functioning, school functioning. A 5-point score is used for each domain: from 0 (never) to 4 (almost always). Items are reversed scored and linearly transformed to a 0-100 scale as follows: 0=100, 1=75, 2=50, 3=25, 4=0 so that higher scores indicate better quality of life (QoL). The total score is the mean (average) of all scores from the 4 domains. The change from baseline in total score is reported- a positive score indicates a better QoL compared to baseline and a negative score indicates a poorer QoL compared to baseline.

Health Resource Use: Unscheduled Doctor's Office VisitsBaseline (Pharmacokinetic [PK] assessment), Week 5, Week 13 and Week 26

The number of unscheduled doctor's Office visits per participant. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \< 6 years of age; pediatric participants 6 to \<12 years of age; Full Analysis Set.

Health Resource Use: Days Lost From SchoolBaseline (Pharmacokinetic [PK] assessment), Week 5, Week 13 and Week 26

The number of days lost from school per participant. Number of participants contributing data (N) for this outcome measure is included in the category title in the order: pediatric participants \< 6 years of age; pediatric participants 6 to \<12 years of age; Full Analysis Set.

Trial Locations

Locations (11)

University Pediatric Hospital

🇵🇱

Krakow, Poland

Stanislaw Popowski Provincial Specialist Pediatric Hospital

🇵🇱

Olsztyn, Poland

Professor Tadeusz Sokolowski Independent Public Teaching Hospital of the Pomeranian Medical University in Szczecin

🇵🇱

Szczecin, Poland

S.C. Sanador SRL

🇷🇴

Bucharest, Romania

Regional Clinical Hospital

🇷🇺

Ekaterinburg, Russian Federation

Pediatric Regional Clinical Hospital, Hematology Department

🇷🇺

Krasnodar, Russian Federation

State Institution "Institute of Blood Pathology and Transfusion Medicine of the Academy of Medical Sciences of Ukraine"

🇺🇦

Lviv, Ukraine

Republican Center for Hemophilia Treatment

🇷🇺

St. Petersburg, Russian Federation

Manchester Children´s Hospital

🇬🇧

Manchester, United Kingdom

Louis Turcanu Emergency Children's Hospital

🇷🇴

Timisoara, Romania

LNJP Maulana Azad Medical College & Associated Hospitals

🇮🇳

New Delhi, India

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