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Safety and Pharmacokinetics of Subcutaneous Injection of OCTA101 in Adult Patients With Severe Hemophilia A

Phase 1
Terminated
Conditions
Severe Hemophilia A
Interventions
Registration Number
NCT04046848
Lead Sponsor
Octapharma
Brief Summary

This Phase 1/2 study will be a dose escalation study in adults in 5 cohorts (named cohorts 1, 2, 3, 5 and 6), with the main purpose to assess the safety of subcutaneous injection of OCTA101 (a human-cl rhFVIII and recombinant human von Willebrand Factor fragment dimer) in previously treated adult patients with severe hemophilia A. The study also aims to assess the pharmacokinetics (PK) characteristics, dose proportionality, and subcutaneous bioavailability of OCTA101 compared with intravenous administration of Nuwiq (Human-cl rh FVIII), in order to define the prophylactic treatment (dose and injection interval) that would result in protective trough levels of FVIII:C for future Phase 3 studies. Cohorts 1, 2, 3 and 5 will undergo a single injection of OCTA101, with cohorts 1, 2 and 3 proceeding to 3-month daily dosing prophylactic treatment for 3 months by Data Monitoring Committee recommendation. Cohorts 1 and 2 will undergo a further PK at the end of the daily injection period. A further cohort, cohort 6, will have an initial 4 to 6-week run-in treatment period with Nuwiq intravenous prophylaxis followed by 12.5 IU/kg OCTA101 subcutaneous daily prophylaxis for \>3 up to 6-7 months.

Detailed Description

Not available

Recruitment & Eligibility

Status
TERMINATED
Sex
Male
Target Recruitment
36
Inclusion Criteria
  1. Severe hemophilia A (<1% FVIII:C) as documented in medical records
  2. Males ≥18 years of age
  3. Subjects who have had ≥150 exposure days (EDs) with a FVIII product
  4. Written informed consent for study participation obtained before undergoing any study specific procedures
Exclusion Criteria
  1. Previous participation in this trial
  2. Use of an Investigational Medicinal Product within 30 days prior to the first OCTA101 injection
  3. History of FVIII inhibitors titre ≥0.6 BU/mL defined by medical records
  4. Inhibitors to FVIII (≥0.6 BU/mL) at screening measured by Nijmegen modified Bethesda method at central laboratory
  5. Human immunodeficiency virus (HIV) positive subjects with a CD4+ count <200/mL
  6. Clinically significant anemia at screening (hemoglobin <8 g/dL)
  7. Presence of any significant comorbidity (at the discretion of the investigator) that might confound the interpretation of the study data and/or that might put the patient at undue risk by participating in the trial
  8. Any coagulation disorder other than hemophilia A
  9. AST or ALT levels >3 times the upper limit of normal
  10. Creatinine >120 μmol/L
  11. Platelet count <100,000 μL
  12. BMI ≥30 kg/m²
  13. For Cohort 6, patients with a positive LumiTope test at screening will be excluded

Study & Design

Study Type
INTERVENTIONAL
Study Design
SEQUENTIAL
Arm && Interventions
GroupInterventionDescription
Cohort 1OCTA10150 IU/kg (n=4): single-period investigation with a single sc dose of 50 IU/kg OCTA101 profiled up to 72 hours after dosing in adult male patients with severe hemophilia A. Following review of safety and tolerability data by Data Monitoring Committee, proceed with Cohort 2, alongside daily prophylactic dosing (40-60 IU/kg) for 3 months.
Cohort 3OCTA10150 IU/kg (n=8): two-period investigation of a single iv dose of 50 IU/kg Human-cl rhFVIII (Nuwiq) profiled for up to 72 hours after dosing followed by sc dose of 50 IU/kg OCTA101 profiled up to 72 hours in adult male patients with severe hemophilia A. Treatments will be administered in fixed sequence, with Human-cl rhFVIII first. Following review of safety and tolerability data by Data Monitoring Committee, proceed with Cohort 4 and 5 alongside daily prophylactic dosing (40-60 IU/kg) for 3 months.
Cohort 5OCTA101(n=4): Three-period investigation of single sc doses of 20, 40, and 60 IU/kg OCTA101 profiled up to 72 hours after dosing. Treatments were to be administered in fixed dose-ascending sequence.
Cohort 6OCTA101(n=16): Following an initial 4 to 6-week run-in period with Nuwiq iv prophylaxis, \>3-6 months daily prophylactic treatment with 12.5 IU/kg OCTA101 sc, then 25 IU/kg OCTA101 sc for a further 6-7 months (exact dosing depends on available vial sizes). In case of two spontaneous bleeding episodes, after having completed at least 3 months with 12.5 IU/kg OCTA101 daily treatment the individual treatment dose will be increased from 12.5 to 25 IU/kg. Site of administration (abdomen or thigh) to be chosen by the patient. A further treatment phase with 40 IU/kg OCTA101 will be discussed with the DMC, once results of earlier dosing phases are available.
Cohort 2OCTA101100 IU/kg (n=4): single-period investigation with a single sc dose of 100 IU/kg OCTA101 profiled up to 96 hours after dosing in adult male patients with severe hemophilia A. Following review of safety and tolerability data by Data Monitoring Committee, proceed with Cohort 3, alongside daily prophylactic dosing (40-60 IU/kg) for 3 months.
Primary Outcome Measures
NameTimeMethod
Patients Experiencing Adverse EventsApproximately 4 months; up to 11 months for cohort 6
Participants Experiencing Dose-limiting Toxicities (DLTs)Approximately 4 months; up to 11 months for cohort 6

Pre-defined DLTs for this study are: 1. Severe allergic reactions at least possibly related to study drug. 2. Severe vital organ toxicity at least possibly related to study drug that does not resolve to at least mild severity within 48 to 72 hours. 3. Any treatment-emergent severe toxicity at least possibly related to study drug other than the toxicities referenced in 2) that does not decrease to mild or resolve within 7 days

Patients Experiencing Thromboembolic EventsApproximately 4 months; up to 11 months for cohort 6

The definition of the cluster thromboembolic events was based on the standardised MedDRA query (SMQ) "Embolic and thrombotic events":

Definition: Thrombotic disorders are diseases characterized by formation of a thrombus that obstructs vascular blood flow locally or detaches and embolizes to occlude blood flow downstream. Embolism is the sudden blocking of a vessel by a clot or foreign material which has been brought to its site of lodgment by the blood current. (Thrombo-)phlebitis is an inflammation of a vein (phlebitis) associated with thrombus formation (thrombosis).

This SMQ includes 3 sub-SMQ:

* Embolic and thrombotic events, venous (SMQ)

* Embolic and thrombotic events, arterial (SMQ)

* Embolic and thrombotic events, vessel type unspecified and mixed arterial and venous (SMQ)

Patients Experiencing Local Injection Site Reactions of Any GradeApproximately 4 months; up to 11 months for cohort 6. Local injection site reactions were captured throughout the period where OCTA-101 was injected subcutaneously (sc).

Investigator (and patient in case of home treatment) assessed local injection reactivity directly after injection and at 15 ± 5 min post-injection as per the ISO10999-10 standard:

0=no skin reactivity;

1. mild (subject is aware of the signs/symptoms, but finds it easily tolerated)

2. moderate (discomfort enough to cause interference with usual activities)

3. severe (subject is incapacitated and unable to work or participate in many or all usual activities).

Inhibitor Formation to FVIIIFrom first injection to 4 months after start of of daily injection (cohorts 1, 2 and 3), 4 weeks after last PK injection (cohort 5), monthly during the daily sc treatment period (cohort 6)

Development of an inhibitor was defined as a neutralizing antibody value of greater than or equal to (\>=) 0.6 Bethesda units per milliliter (BU/mL) identified and confirmed by a second test on an independent sample. In case of positive inhibitor results, inhibitor retesting using a second, separately drawn sample was to be performed, preferably within 15 days of becoming aware of the positive result. Both tests performed by the central laboratory using Nijmegen-modified Bethesda assay.

Secondary Outcome Measures
NameTimeMethod
Efficacy: Area Under the Concentration-time Curve (AUC) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Mean AUC of FVIII after PK injection of OCTA101 as measured by chromogenic assay.

Efficacy: Maximum Plasma Concentration (Cmax) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Maximum observed concentration of FVIII:C after PK injection of OCTA101 as measured by chromogenic assay.

Efficacy: Time for Reaching Maximum Plasma Concentration (Tmax) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Time of occurrence of Cmax after PK injection of OCTA101 as measured by chromogenic assay.

In Vivo Recovery (IVR) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

In vivo recovery (IVR) = dose-normalised and body weight-normalised maximum gain in FVIII:C (IU/dL per IU/kg)

Efficacy: Half-life (t1/2) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Apparent terminal log-linear half-life of FVIII:C after PK injection of OCTA101 as measured by chromogenic assay.

Efficacy: Mean Residence Time (MRT) of FVIII:CFrom 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

The average time at which the number of absorbed FVIII molecules reside in the body, after PK injection of OCTA101 as measured by chromogenic assay.

Efficacy: Area Under the Concentration-time Curve (AUC(0-tz)) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Measurement of OCTA12 plasma concentrations using a validated ELISA in a central lab. Note: OCTA12 was not determined for cohort 6 regarding PK assessment.

Efficacy: Maximum Plasma Concentration (Cmax) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Measurement of OCTA12 plasma concentrations using a validated ELISA in a central lab. Note: OCTA12 was not determined for cohort 6 regarding PK assessment.

Efficacy: Time for Reaching Maximum Plasma Concentration (Tmax) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Measurement of OCTA12 plasma concentrations using a validated ELISA in a central lab. Note: OCTA12 was not determined for cohort 6 regarding PK assessment.

Efficacy: In Vivo Recovery (IVR) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

In vivo recovery (IVR) = dose-normalised and body weight-normalised maximum gain in OCTA-12 (ug/dL per ug/kg).

Efficacy: Half Life (t1/2) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Measurement of OCTA12 plasma concentrations using a validated ELISA in a central lab. Note: t(1/2) could not be determined for OTCA12 as the OCTA12 concentrations had not yet declined during the observation time prior to prior to end of sampling period.

Efficacy: Mean Residence Time (MRT) of OCTA12 (a Recombinant Von Willebrand Factor Fragment Dimer)From 0 hours (pre-dose) to 72 hours (cohorts 1,3,5) or 96 hours (cohort 2)

Note: MRT could not be determined for OCTA12 as the OCTA12 concentrations had not yet declined during the observation time prior to end of sampling period.

Efficacy: Total Annualized Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93).

Total annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3 An estimated total annualized bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate for cohort 6.

Efficacy: Spontaneous Annualized Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93)

Spontaneous annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3

An estimated total spontaneous annualized bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate

Efficacy: Total Annualized Treated Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93)

Total annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3

An estimated total annualized treated bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate.

Efficacy: Spontaneous Annualized Treated Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93)

Spontaneous annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3.

An estimated total spontaneous annualized treated bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate.

Efficacy: Traumatic Annualized Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93)

Traumatic annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3

An estimated total traumatic annualized bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate.

Efficacy: Joint Annualized Bleeding RateThe median (min, max) duration of daily sc treatment with OCTA101 in the 16 patients of cohort 1, 2 and 3 was 42 days (22, 93)

Joint annualized bleeding rate during daily subcutaneous treatment with OCTA101 for cohorts 1, 2 and 3.

An estimated total joint annualized bleeding rate was calculated for cohorts 1,2 and 3. As these were estimated rates, there is only one value for each cohort with no measure of spread.

As the study was terminated so soon, it was not considered accurate to extrapolate results to one year for annualized bleeding rate.

Efficacy: FVIII:C Trough and Peak Plasma Levels3 months; maximally 6 months for cohort 6

FVIII:C trough and peak plasma levels during daily dosing for cohorts 1, 2, 3 and 6

Efficacy: Efficacy of Treatment of Bleeding Episodes Using Score (4-point).5 days to approximately 11 months

Score (4-point) to assess the efficacy of treatment of bleeding episodes with Human-cl rhFVIII. Treatment efficacy will be assessed using predefined criteria to score either 'Excellent', 'Good', 'Moderate' or 'None'. All efficacy ratings assessed as either 'excellent' or 'good' will be considered 'successfully treated'.

Safety: Antibody Formation to OCTA12From 0 hours (pre-dose) to 72 hours (cohorts 1,2,3,5) or 96 hours (cohort 2).

Samples were checked for the presence of antibodies to OCTA12 by using a validated ELISA in a central lab.

Safety: OCTA12 Plasma Levels3 months; maximally 6 months for cohort 6

OCTA12 plasma levels during daily dosing (cohorts 1, 2, 3 and 6).

Safety: Change in Hemoglobin5 days to approximately 11 months. All routine lab parameters and vital signs were measured at various times, until end of PK and also monthly during daily prophylaxis.)

Number of Participants with changes in hemoglobin levels that were considered as Adverse Events.

Safety: Change in Alanine Aminotransferase (ALT)5 days to approximately 11 months. All routine lab parameters and vital signs were measured at various times, until end of PK and also monthly during daily prophylaxis.)

Alanine aminotransferase (ALT) compared to baseline, measured in U/L. Number of Participants with changes in ALT that were considered as Adverse Events.

Safety: Change in Aspartate Transaminase (AST)5 days to approximately 11 months

Aspartate transaminase (AST) compared to baseline, measured in U/l. Number of Participants with changes in AST that were considered as Adverse Events.

Patients With Changes to Vital Signs5 days to approximately 11 months

Vitals signs changes from baseline, reported as AEs. Number of Participants with changes to vital signs that were considered as Adverse Events.

Patients With Changes in Physical Examination Results5 days to approximately 11 months

Number of Participants with changes to their physical examination results from baseline that were considered as Adverse Events

Trial Locations

Locations (1)

Specialized Hospital for Active Treatment of Hematological Diseases EAD Clinic of Clinical Hematology

🇧🇬

Sofia, Bulgaria

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