Trial Outcomes & Findings for Long-term Safety of Alpha1-Proteinase Inhibitor (Human) in Japanese Subjects With Alpha1 Antitrypsin Deficiency (GTI1401-OLE) (NCT NCT02870348)

NCT ID: NCT02870348

Last Updated: 2022-03-31

Results Overview

An adverse event (AE) was defined as any untoward medical occurrence in a participant or clinical investigation participant administered a medicinal product or study treatment and which did not necessarily have a causal relationship with this administration. TEAEs were defined as any AE occurring after or on the first Alpha-1 MP infusion until the final visit of study.

Recruitment status

COMPLETED

Study phase

PHASE1/PHASE2

Target enrollment

4 participants

Primary outcome timeframe

From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Results posted on

2022-03-31

Participant Flow

The study was conducted at 3 investigative sites in Japan from 29 July 2016 to 16 February 2021.

Participants with diagnosis of alpha1-antitrypsin deficiency (AATD) who completed the Study GTI1401 (NCT02870309) were eligible to enroll in this open-label extension study. The Screening / Extension (Ext) Week 1 Visit and initiation of treatment for the current study (GTI1401-OLE) occurred on the same day as the Week 9 Visit of GTI1401 only for those participants who gave the consent for participation in the Study GTI1401-OLE.

Participant milestones

Participant milestones
Measure
Alpha-1 MP
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Overall Study
STARTED
4
Overall Study
COMPLETED
0
Overall Study
NOT COMPLETED
4

Reasons for withdrawal

Reasons for withdrawal
Measure
Alpha-1 MP
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Overall Study
Participants were discontinued and assessed for entry into post-marketing study (GTI1401-EXP)
4

Baseline Characteristics

Long-term Safety of Alpha1-Proteinase Inhibitor (Human) in Japanese Subjects With Alpha1 Antitrypsin Deficiency (GTI1401-OLE)

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Age, Continuous
60.8 years
STANDARD_DEVIATION 5.56 • n=5 Participants
Sex: Female, Male
Female
1 Participants
n=5 Participants
Sex: Female, Male
Male
3 Participants
n=5 Participants
Race (NIH/OMB)
American Indian or Alaska Native
0 Participants
n=5 Participants
Race (NIH/OMB)
Asian
4 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
0 Participants
n=5 Participants
Race (NIH/OMB)
More than one race
0 Participants
n=5 Participants
Race (NIH/OMB)
Unknown or Not Reported
0 Participants
n=5 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

An adverse event (AE) was defined as any untoward medical occurrence in a participant or clinical investigation participant administered a medicinal product or study treatment and which did not necessarily have a causal relationship with this administration. TEAEs were defined as any AE occurring after or on the first Alpha-1 MP infusion until the final visit of study.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Treatment-Emergent Adverse Events (TEAEs)
4 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

ADRs were defined as all noxious and unintended responses to a medicinal product or study treatment related to any dose. An AE was defined as any untoward medical occurrence in a participant or clinical investigation participant administered a medicinal product or study treatment and which did not necessarily have a causal relationship with this administration.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Adverse Drug Reaction (ADRs)
2 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

An AE was considered serious in any of the following outcomes or deemed significant for any other reason: death; life-threatening AE; inpatient hospitalization or prolongation of existing hospitalization; persistent or significant incapacity or substantial disruption of the ability to conduct normal life functions; congenital anomaly/birth defect.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Serious Adverse Events (SAEs)
2 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

An AE was defined as any untoward medical occurrence in a participant or clinical investigation participant administered a medicinal product or study treatment and which did not necessarily have a causal relationship with this administration. An AE was considered serious in any of the following outcomes or deemed significant for any other reason: death; life-threatening AE; inpatient hospitalization or prolongation of existing hospitalization; persistent or significant incapacity or substantial disruption of the ability to conduct normal life functions; congenital anomaly/birth defect.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Discontinuations Due to Adverse Events (AEs) or Serious Adverse Events (SAEs)
0 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

COPD exacerbation was defined as an increase in respiratory symptoms (dyspnea, increased cough, and/or production of sputum) over baseline that usually requires medical intervention.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Chronic Obstructive Pulmonary Disease (COPD) Exacerbations
0 Participants

PRIMARY outcome

Timeframe: From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

Vital signs included analysis of heart rate, blood pressure, respiratory rate, and temperature. Clinically significant findings in vital signs were based on investigator's discretion.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Clinically Significant Findings in Vital Signs
0 Participants

PRIMARY outcome

Timeframe: Extension (Ext) Weeks 26, 52, 78, 104, 130, 156,182, 208 (prior to study drug administration) until end of study (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

Clinical laboratory parameters included analysis of hematology, blood chemistry, and urinalysis. Clinically significant findings in clinical laboratory parameters were based on investigator's discretion.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Clinically Significant Findings in Clinical Laboratory Parameters
0 Participants

PRIMARY outcome

Timeframe: Extension (Ext) Weeks 26, 52, 78, 104, 130, 156,182, 208 (prior to study drug administration) until end of study (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

Pulmonary function tests were measured by spirometry. It included Forced Expired Volume in 1 second (FEV1), Forced Vital Capacity (FVC), which were performed according to American Thoracic Society/ European Respiratory Society (ATS/ERS) guidelines. FEV1 is the amount of air which can be forcibly exhaled from the lungs in the first second of a forced exhalation. FVC is the amount of air which can be forcibly exhaled from the lungs after taking the deepest breath possible. PFTs were performed before infusion both pre- and post-bronchodilator administration. The post-bronchodilator PFT was performed 15 to 30 minutes after bronchodilator administration. The same bronchodilator was used throughout the study. Clinically significant findings in pulmonary function tests were based on investigator's discretion.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Number of Participants With Clinically Significant Findings in Pulmonary Function Tests (PFTs)
0 Participants

PRIMARY outcome

Timeframe: Extension (Ext) Weeks 12, 24, 36, 48, 64, 76, 88, 100, 116, 128, 140, 152, 168, 180, 192, 204 (prior to study drug administration) until end of study (Up to 228 weeks)

Population: Safety Population included all participants who received any amount of Alpha-1 MP.

Alpha1-PI level was measured by nephelometry.

Outcome measures

Outcome measures
Measure
Alpha-1 MP
n=4 Participants
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Trough Levels of Alpha1- Proteinase Inhibitor
55.734 milligram/deciliter (mg/dL)
Standard Deviation 4.9928

Adverse Events

Alpha-1 MP

Serious events: 2 serious events
Other events: 4 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
Alpha-1 MP
n=4 participants at risk
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Respiratory, thoracic and mediastinal disorders
Pulmonary hypertension
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Pneumonia
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Cellulitis
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Large intestine polyp
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Pneumothorax
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.

Other adverse events

Other adverse events
Measure
Alpha-1 MP
n=4 participants at risk
Participants received IV infusion of 60 mg/kg Alpha-1 MP administered weekly for mean duration of 213 weeks.
Cardiac disorders
Arrhythmia
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Cardiac disorders
Palpitations
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Eye disorders
Conjunctival haemorrhage
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Eye disorders
Eyelid pain
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Eye disorders
Vitreous floaters
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Abdominal discomfort
50.0%
2/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Abdominal pain
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Diarrhoea
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Faeces soft
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Gastritis
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Gastrointestinal disorder
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Gastrooesophageal reflux disease
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Gastrointestinal disorders
Haemorrhoids
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Hepatobiliary disorders
Hepatic function abnormal
50.0%
2/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Gastroenteritis
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Nasopharyngitis
50.0%
2/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Pharyngitis
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Injury, poisoning and procedural complications
Arthropod sting
50.0%
2/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Investigations
Blood creatinine increased
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Investigations
Influenza virus test positive
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Metabolism and nutrition disorders
Gout
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Musculoskeletal and connective tissue disorders
Arthralgia
50.0%
2/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Musculoskeletal and connective tissue disorders
Back pain
75.0%
3/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Cough
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Epistaxis
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Pulmonary mass
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Rhinorrhoea
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Respiratory, thoracic and mediastinal disorders
Upper respiratory tract inflammation
100.0%
4/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Skin and subcutaneous tissue disorders
Erythema
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Vascular disorders
Hot flush
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.
Infections and infestations
Pneumonia
25.0%
1/4 • From start of study drug administration through 30 days after last study drug infusion (Up to 228 weeks)
Safety Population included all participants who received any amount of Alpha-1 MP.

Additional Information

Sandra Camprubi

Instituto Grifols SA

Phone: +34935710049

Results disclosure agreements

  • Principal investigator is a sponsor employee Site may publish results from the Study, after providing Sponsor thirty days' notice prior to submitting a manuscript or other materials related to the Study to any outside party. At Sponsors' request, Site will remove any Confidential Information (other than Study results), and Site will upon Sponsors' request, delay publication or presentation for a period of up to one hundred twenty days to allow Sponsor to protect its interests in any Sponsor Inventions.
  • Publication restrictions are in place

Restriction type: OTHER