Trial Outcomes & Findings for Dose-finding Study of SPK-8016 Gene Therapy in Patients With Hemophilia A to Support Evaluation in Individuals With FVIII Inhibitors (NCT NCT03734588)

NCT ID: NCT03734588

Last Updated: 2024-02-23

Results Overview

An AE was defined as any untoward medical occurrence in a participant who received study drug without regard to possibility of causal relationship. SAEs were defined as death, a life-threatening AE, inpatient hospitalization or prolongation of existing hospitalization, persistent or significant disability or incapacity, a congenital anomaly or birth defect, or an important medical event that jeopardized participant and required medical intervention to prevent 1 of the outcomes listed in this definition. A summary of other non-serious AEs and all serious AEs, regardless of causality is located in Reported AE section.

Recruitment status

COMPLETED

Study phase

PHASE1/PHASE2

Target enrollment

4 participants

Primary outcome timeframe

Up to week 52

Results posted on

2024-02-23

Participant Flow

A dose-finding part of this trial was planned but no participants were enrolled into the higher dose arms. All participants received SPK-8016 5x10\^11 vg/kg

Participant milestones

Participant milestones
Measure
SPK-8016
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vector genomes per kilogram body weight (vg/kg).
Overall Study
STARTED
4
Overall Study
COMPLETED
4
Overall Study
NOT COMPLETED
0

Reasons for withdrawal

Withdrawal data not reported

Baseline Characteristics

Dose-finding Study of SPK-8016 Gene Therapy in Patients With Hemophilia A to Support Evaluation in Individuals With FVIII Inhibitors

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Age, Continuous
36.3 years
STANDARD_DEVIATION 19.29 • n=99 Participants
Sex: Female, Male
Female
0 Participants
n=99 Participants
Sex: Female, Male
Male
4 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Hispanic or Latino
0 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Not Hispanic or Latino
4 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Unknown or Not Reported
0 Participants
n=99 Participants
Race (NIH/OMB)
American Indian or Alaska Native
0 Participants
n=99 Participants
Race (NIH/OMB)
Asian
0 Participants
n=99 Participants
Race (NIH/OMB)
Native Hawaiian or Other Pacific Islander
0 Participants
n=99 Participants
Race (NIH/OMB)
Black or African American
0 Participants
n=99 Participants
Race (NIH/OMB)
White
4 Participants
n=99 Participants
Race (NIH/OMB)
More than one race
0 Participants
n=99 Participants
Race (NIH/OMB)
Unknown or Not Reported
0 Participants
n=99 Participants

PRIMARY outcome

Timeframe: Up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

An AE was defined as any untoward medical occurrence in a participant who received study drug without regard to possibility of causal relationship. SAEs were defined as death, a life-threatening AE, inpatient hospitalization or prolongation of existing hospitalization, persistent or significant disability or incapacity, a congenital anomaly or birth defect, or an important medical event that jeopardized participant and required medical intervention to prevent 1 of the outcomes listed in this definition. A summary of other non-serious AEs and all serious AEs, regardless of causality is located in Reported AE section.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Number of Participants With Adverse Events (AEs)
4 Participants

PRIMARY outcome

Timeframe: Up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Number of Participants With Hepatic Transaminase Elevation Requiring Immunosuppression.
0 Participants

PRIMARY outcome

Timeframe: Up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Peak FVIII Activity Levels Assessed by Coagulation Clotting Assays
NA percentage of normal activity
Data was not analyzed due to low number of participants enrolled. Data values are not reported due to privacy concerns related to low enrollment.

PRIMARY outcome

Timeframe: Up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Steady-state FVIII Activity Levels Assessed by Coagulation Clotting Assays
NA percentage of normal activity
Standard Deviation NA
Insufficient number of participants with events

PRIMARY outcome

Timeframe: From 28 days post vector administration up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Number of Bleeding Events (Spontaneous and Traumatic) Since 28 Day Post Vector Administration
Traumatic
6 Number of bleeding events
Number of Bleeding Events (Spontaneous and Traumatic) Since 28 Day Post Vector Administration
Spontaneous
1 Number of bleeding events

PRIMARY outcome

Timeframe: From 28 days post vector administration up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Annualized Infusion Rate
NA Annualized Infusion Rate
Data was not analyzed due to low number of participants enrolled. Data values are not reported due to privacy concerns related to low enrollment.

SECONDARY outcome

Timeframe: Up to week 52

Population: The FAS included all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Time to Achieve Steady-state FVIII Activity Levels
NA hours
Standard Deviation NA
Insufficient number of participants with events

SECONDARY outcome

Timeframe: Up to week 52

Population: Results were uninterpretable due to a technical error.

Outcome measures

Outcome data not reported

SECONDARY outcome

Timeframe: Up to week 52

Population: The FAS includes all enrolled participants who received the infusion of SPK-8016.

Outcome measures

Outcome measures
Measure
SPK-8016
n=4 Participants
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg.
Number of Participants With Immune Responses to AAV Capsid Protein and BDD-hFVIII Transgene
3 Participants

Adverse Events

SPK-8016

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

Serious adverse events

Serious adverse events
Measure
SPK-8016
n=4 participants at risk
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg
Gastrointestinal disorders
Food Poisoning
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Haematemesis
25.0%
1/4 • Up to week 52
Musculoskeletal and connective tissue disorders
Back Pain
25.0%
1/4 • Up to week 52

Other adverse events

Other adverse events
Measure
SPK-8016
n=4 participants at risk
Participants received a single intravenous infusion of SPK-8016 at 5x10\^11 vg/kg
Endocrine disorders
Cushingoid
25.0%
1/4 • Up to week 52
Eye disorders
Vision Blurred
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Abdominal Pain Upper
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Diarrhoea
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Dyspepsia
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Food Poisoning
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Nausea
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Tooth Loss
25.0%
1/4 • Up to week 52
Gastrointestinal disorders
Vomiting
25.0%
1/4 • Up to week 52
General disorders
Fatigue
25.0%
1/4 • Up to week 52
General disorders
Feeling Hot
25.0%
1/4 • Up to week 52
General disorders
Oedema Peripheral
25.0%
1/4 • Up to week 52
General disorders
Swelling Face
25.0%
1/4 • Up to week 52
Hepatobiliary disorders
Drug-Induced Liver Injury
25.0%
1/4 • Up to week 52
Infections and infestations
Candida Infection
25.0%
1/4 • Up to week 52
Infections and infestations
Gastroenteritis Viral
25.0%
1/4 • Up to week 52
Infections and infestations
Nasopharyngitis
25.0%
1/4 • Up to week 52
Infections and infestations
Oral Herpes
25.0%
1/4 • Up to week 52
Infections and infestations
Pharyngitis Streptococcal
25.0%
1/4 • Up to week 52
Infections and infestations
Skin Infection
25.0%
1/4 • Up to week 52
Infections and infestations
Tooth Abscess
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Contusion
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Limb Crushing Injury
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Scratch
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Skin Abrasion
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Skin Laceration
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Thermal Burn
25.0%
1/4 • Up to week 52
Injury, poisoning and procedural complications
Tooth Fracture
25.0%
1/4 • Up to week 52
Investigations
Blood Glucose Increased
25.0%
1/4 • Up to week 52
Investigations
Weight Increased
50.0%
2/4 • Up to week 52
Metabolism and nutrition disorders
Hyperglycaemia
25.0%
1/4 • Up to week 52
Musculoskeletal and connective tissue disorders
Arthralgia
50.0%
2/4 • Up to week 52
Musculoskeletal and connective tissue disorders
Joint Noise
25.0%
1/4 • Up to week 52
Musculoskeletal and connective tissue disorders
Joint Swelling
25.0%
1/4 • Up to week 52
Musculoskeletal and connective tissue disorders
Muscular Weakness
25.0%
1/4 • Up to week 52
Nervous system disorders
Dizziness
25.0%
1/4 • Up to week 52
Nervous system disorders
Headache
50.0%
2/4 • Up to week 52
Nervous system disorders
Hypoaesthesia
25.0%
1/4 • Up to week 52
Nervous system disorders
Lethargy
50.0%
2/4 • Up to week 52
Psychiatric disorders
Anxiety
25.0%
1/4 • Up to week 52
Psychiatric disorders
Libido Decreased
25.0%
1/4 • Up to week 52
Renal and urinary disorders
Pollakiuria
25.0%
1/4 • Up to week 52
Respiratory, thoracic and mediastinal disorders
Cough
25.0%
1/4 • Up to week 52
Respiratory, thoracic and mediastinal disorders
Oropharyngeal Pain
50.0%
2/4 • Up to week 52
Respiratory, thoracic and mediastinal disorders
Pharyngeal Erythema
25.0%
1/4 • Up to week 52
Respiratory, thoracic and mediastinal disorders
Wheezing
25.0%
1/4 • Up to week 52
Skin and subcutaneous tissue disorders
Acne
25.0%
1/4 • Up to week 52
Skin and subcutaneous tissue disorders
Pruritus
25.0%
1/4 • Up to week 52
Skin and subcutaneous tissue disorders
Rash
25.0%
1/4 • Up to week 52
Skin and subcutaneous tissue disorders
Skin Irritation
25.0%
1/4 • Up to week 52

Additional Information

Tiffany Chang, MD, MAS Clinical Development Lead, Hematology

Spark Therapeutics

Phone: Please Email

Results disclosure agreements

  • Principal investigator is a sponsor employee
  • Publication restrictions are in place