Trial Outcomes & Findings for Study to Investigate Immunogenicity, Efficacy and Safety of Treatment With Human-cl rhFVIII (NCT NCT01992549)

NCT ID: NCT01992549

Last Updated: 2021-01-19

Results Overview

The number of patients developing FVIII inhibitors was observed during the observation period by assessing inhibitor development by the modified Bethesda assay (Nijmegen modification) using congenital FVIII-deficient human plasma spiked with Human-cl rhFVIII. The definition threshold for a "positive" inhibitor was if the modified Bethesda assay resulted in a titre ≥0.6 BU/mL at any time point during the observation period.

Recruitment status

COMPLETED

Study phase

PHASE3

Target enrollment

48 participants

Primary outcome timeframe

Maximum two years

Results posted on

2021-01-19

Participant Flow

Participant milestones

Participant milestones
Measure
Human-cl rhFVIII
Of the total number of patients that started in the study, all those in the safety (SAF) population received at least 1 infusion of Human-cl rhFVIII. Patients who had data collected post-treatment were included in the intent-to-treat (ITT) population. Prophylactic treatment dose given to all patients in the PROPH population (all patients who received at least 1 administration of Human-cl rhFVIII with prophylaxis documented as the reason for treatment): \>20 IU FVIII/kg body weight (BW). On-demand treatment of bleeding episodes (BEs) dose given to the BLEED population (all patients with bleeding episodes treated with Human-cl rhFVIII): 20-30 IU FVIII/kg BW (minor haemorrhage), 30-40 IU FVIII/kg BW (moderate to major haemorrhage) or 50-80 IU FVIII/kg BW (major to life-threatening haemorrhage). Surgical prophylaxis dose was given to the SURG population (all patients with surgeries treated with Human-cl rhFVIII): 25-30 IU FVIII/kg BW (minor surgeries); \>50 IU FVIII/kg BW (major surgeries).
Overall Study
STARTED
48
Overall Study
SAF Population
48
Overall Study
ITT Population
47
Overall Study
PROPH Population
47
Overall Study
BLEED Population
29
Overall Study
SURG Population
3
Overall Study
COMPLETED
44
Overall Study
NOT COMPLETED
4

Reasons for withdrawal

Reasons for withdrawal
Measure
Human-cl rhFVIII
Of the total number of patients that started in the study, all those in the safety (SAF) population received at least 1 infusion of Human-cl rhFVIII. Patients who had data collected post-treatment were included in the intent-to-treat (ITT) population. Prophylactic treatment dose given to all patients in the PROPH population (all patients who received at least 1 administration of Human-cl rhFVIII with prophylaxis documented as the reason for treatment): \>20 IU FVIII/kg body weight (BW). On-demand treatment of bleeding episodes (BEs) dose given to the BLEED population (all patients with bleeding episodes treated with Human-cl rhFVIII): 20-30 IU FVIII/kg BW (minor haemorrhage), 30-40 IU FVIII/kg BW (moderate to major haemorrhage) or 50-80 IU FVIII/kg BW (major to life-threatening haemorrhage). Surgical prophylaxis dose was given to the SURG population (all patients with surgeries treated with Human-cl rhFVIII): 25-30 IU FVIII/kg BW (minor surgeries); \>50 IU FVIII/kg BW (major surgeries).
Overall Study
Lost to Follow-up
2
Overall Study
Serious Adverse Event
1
Overall Study
Product Switch to Extend Half Life
1

Baseline Characteristics

Study to Investigate Immunogenicity, Efficacy and Safety of Treatment With Human-cl rhFVIII

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Human-cl rhFVIII
n=48 Participants
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII (n=48).
Age, Continuous
3.47 Years
STANDARD_DEVIATION 2.13 • n=5 Participants
Sex: Female, Male
Female
0 Participants
n=5 Participants
Sex: Female, Male
Male
48 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Hispanic or Latino
0 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Not Hispanic or Latino
48 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Unknown or Not Reported
0 Participants
n=5 Participants
Race (NIH/OMB)
American Indian or Alaska Native
1 Participants
n=5 Participants
Race (NIH/OMB)
Asian
9 Participants
n=5 Participants
Race (NIH/OMB)
Native Hawaiian or Other Pacific Islander
0 Participants
n=5 Participants
Race (NIH/OMB)
Black or African American
0 Participants
n=5 Participants
Race (NIH/OMB)
White
36 Participants
n=5 Participants
Race (NIH/OMB)
More than one race
2 Participants
n=5 Participants
Race (NIH/OMB)
Unknown or Not Reported
0 Participants
n=5 Participants
Body Mass Index (BMI) at screening
16.46 kg/m^2
n=5 Participants
Height at screening
97.44 Centimeters (cm)
n=5 Participants
Weight at screening
15.84 Kilograms (kg)
n=5 Participants
Family history of inhibitors
Yes
3 Participants
n=5 Participants
Family history of inhibitors
No
45 Participants
n=5 Participants

PRIMARY outcome

Timeframe: Maximum two years

Population: The analysis was performed for the safety (SAF) population which includes all patients who received at least 1 infusion of Human-cl rhFVIII (N=48)

The number of patients developing FVIII inhibitors was observed during the observation period by assessing inhibitor development by the modified Bethesda assay (Nijmegen modification) using congenital FVIII-deficient human plasma spiked with Human-cl rhFVIII. The definition threshold for a "positive" inhibitor was if the modified Bethesda assay resulted in a titre ≥0.6 BU/mL at any time point during the observation period.

Outcome measures

Outcome measures
Measure
Human-cl rhFVIII
n=48 Participants
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII
Major Surgeries
All patients that had major surgeries performed under Human-cl rhFVIII treatment (n=2)
SURG Population
All patients that had surgeries performed under Human-cl rhFVIII treatment (n=3)
Immunogenicity of Human-cl rhFVIII: Incidence of Inhibitors
0 participants
Interval 0.0 to 7.549

SECONDARY outcome

Timeframe: Maximum 2 years

Population: The analysis population includes all patients in PROPH population who received at least one prophylactic treatment with Human-cl rhFVIII (n=47).

The annualized bleeding rate (ABR) was calculated during the time of prophylactic treatment with Human-cl rhFVIII for spontaneous bleeding events (BEs).

Outcome measures

Outcome measures
Measure
Human-cl rhFVIII
n=47 Participants
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII
Major Surgeries
All patients that had major surgeries performed under Human-cl rhFVIII treatment (n=2)
SURG Population
All patients that had surgeries performed under Human-cl rhFVIII treatment (n=3)
Frequency of Spontaneous Break-through Bleeds
0.283 Events/year (ABR)
Interval 0.153 to 0.527

SECONDARY outcome

Timeframe: Maximum 2 years

Population: The analysis population included all patients who received on-demand treatment with Human-cl rhFVIII for bleeding episodes (BLEED population; n=29).

A personal efficacy assessment (final outcome) to assess the efficacy of Human-cl rhFVIII for the on-demand treatment of bleeding episodes (BEs) at the end of a BE. Efficacy was assessed using a four-point scale (excellent, good, moderate, none) by the patient's parent(s)/legal guardian(s) together with the investigator in case of on site treatment.

Outcome measures

Outcome measures
Measure
Human-cl rhFVIII
n=111 Bleeding episodes
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII
Major Surgeries
All patients that had major surgeries performed under Human-cl rhFVIII treatment (n=2)
SURG Population
All patients that had surgeries performed under Human-cl rhFVIII treatment (n=3)
Efficacy of Human-cl rhFVIII for the Treatment of Bleeds
Excellent
58 Bleeding episodes
Efficacy of Human-cl rhFVIII for the Treatment of Bleeds
Good
28 Bleeding episodes
Efficacy of Human-cl rhFVIII for the Treatment of Bleeds
Moderate
21 Bleeding episodes
Efficacy of Human-cl rhFVIII for the Treatment of Bleeds
None
3 Bleeding episodes
Efficacy of Human-cl rhFVIII for the Treatment of Bleeds
Not done
1 Bleeding episodes

SECONDARY outcome

Timeframe: Maximum 2 years

Population: The analysis population includes 3 patients that received Human-cl rhFVIII for surgical prophylaxis during a total of 4 surgeries (SURG population). Of these, 1 patient had two minor surgeries and 2 patients had one major surgery each.

An overall efficacy assessment to assess the efficacy of human-cl rhFVIII in surgical prophylaxis of minor and major surgeries. The efficacy assessment was analyzed using a four-point scale (excellent, good, moderate, none). If surgeries could not be assessed due to limited data available or having taken place outside the study site, the results were classified as "not done".

Outcome measures

Outcome measures
Measure
Human-cl rhFVIII
n=2 Surgeries
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII
Major Surgeries
n=2 Surgeries
All patients that had major surgeries performed under Human-cl rhFVIII treatment (n=2)
SURG Population
n=4 Surgeries
All patients that had surgeries performed under Human-cl rhFVIII treatment (n=3)
Efficacy of Human-cl rhFVIII for Surgical Prophylaxis
Good
0 Surgeries
0 Surgeries
0 Surgeries
Efficacy of Human-cl rhFVIII for Surgical Prophylaxis
Not done
2 Surgeries
0 Surgeries
2 Surgeries
Efficacy of Human-cl rhFVIII for Surgical Prophylaxis
Excellent
0 Surgeries
2 Surgeries
2 Surgeries
Efficacy of Human-cl rhFVIII for Surgical Prophylaxis
Moderate
0 Surgeries
0 Surgeries
0 Surgeries
Efficacy of Human-cl rhFVIII for Surgical Prophylaxis
None
0 Surgeries
0 Surgeries
0 Surgeries

SECONDARY outcome

Timeframe: Maximum 2 years

Population: The analysis was performed for the safety (SAF) population which includes all patients who received at least 1 infusion of Human-cl rhFVIII (n=48).

The frequency of AEs, as monitored throughout the whole study by the number of patients with at least one adverse event occurrence.

Outcome measures

Outcome measures
Measure
Human-cl rhFVIII
n=48 Participants
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII
Major Surgeries
All patients that had major surgeries performed under Human-cl rhFVIII treatment (n=2)
SURG Population
All patients that had surgeries performed under Human-cl rhFVIII treatment (n=3)
The Occurrence of Any Adverse Event (AE)
Adverse event
29 Participants
The Occurrence of Any Adverse Event (AE)
Serious adverse event (SAE)
5 Participants
The Occurrence of Any Adverse Event (AE)
Severe AE
3 Participants
The Occurrence of Any Adverse Event (AE)
Temporally related adverse event
20 Participants
The Occurrence of Any Adverse Event (AE)
Death
0 Participants
The Occurrence of Any Adverse Event (AE)
AE leading to permanent discontinuation
1 Participants

Adverse Events

SAF Population

Serious events: 5 serious events
Other events: 29 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
SAF Population
n=48 participants at risk
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII (n=48).
Infections and infestations
Pneumonia
4.2%
2/48 • Maximum 2 years
Infections and infestations
Varicella
2.1%
1/48 • Maximum 2 years
Respiratory, thoracic and mediastinal disorders
Epistaxis
2.1%
1/48 • Maximum 2 years
Vascular disorders
Haemorrhage
2.1%
1/48 • Maximum 2 years
Gastrointestinal disorders
Gastritis
2.1%
1/48 • Maximum 2 years
Infections and infestations
Cellulitis
2.1%
1/48 • Maximum 2 years
Neoplasms benign, malignant and unspecified (incl cysts and polyps)
Neuroblastoma
2.1%
1/48 • Maximum 2 years

Other adverse events

Other adverse events
Measure
SAF Population
n=48 participants at risk
The safety (SAF) population consist of all patients who received at least one infusion of Human-cl rhFVIII (n=48).
Infections and infestations
Nasopharyngitis
20.8%
10/48 • Maximum 2 years
Infections and infestations
Varicella
10.4%
5/48 • Maximum 2 years
Infections and infestations
Bronchitis
8.3%
4/48 • Maximum 2 years
Infections and infestations
Ear infection
8.3%
4/48 • Maximum 2 years
Infections and infestations
Pharyngitis
8.3%
4/48 • Maximum 2 years
Infections and infestations
Tonsillitis
6.2%
3/48 • Maximum 2 years
Infections and infestations
Otitis media
4.2%
2/48 • Maximum 2 years
Infections and infestations
Pneumonia
4.2%
2/48 • Maximum 2 years
Infections and infestations
Pulpitis dental
4.2%
2/48 • Maximum 2 years
Infections and infestations
Respiratory tract infection viral
4.2%
2/48 • Maximum 2 years
Infections and infestations
Tracheitis
4.2%
2/48 • Maximum 2 years
General disorders
Pyrexia
20.8%
10/48 • Maximum 2 years
Respiratory, thoracic and mediastinal disorders
Cough
14.6%
7/48 • Maximum 2 years
Respiratory, thoracic and mediastinal disorders
Rhinorrhoea
4.2%
2/48 • Maximum 2 years
Gastrointestinal disorders
Diarrhoea
4.2%
2/48 • Maximum 2 years
Gastrointestinal disorders
Vomiting
4.2%
2/48 • Maximum 2 years
Blood and lymphatic system disorders
Anaemia
8.3%
4/48 • Maximum 2 years

Additional Information

Sylvia Werner

Octapharma

Phone: 415 260-9577

Results disclosure agreements

  • Principal investigator is a sponsor employee Octapharma agreements may vary with individual investigators, but will not prohibit any investigator from publishing. Octapharma supports the publication of results from all centers of a multi-center trial but requests that reports based on single-site data not precede the primary publication of the entire clinical trial. Octapharma also reserves the right to review data prior to publishing and provide comments/changes within a certain time period.
  • Publication restrictions are in place

Restriction type: OTHER