Trial Outcomes & Findings for Phase 3 Study of Cysteamine Bitartrate Delayed-release (RP103) Compared to Cystagon® in Patients With Cystinosis (NCT NCT01000961)

NCT ID: NCT01000961

Last Updated: 2024-12-19

Results Overview

Recruitment status

COMPLETED

Study phase

PHASE3

Target enrollment

43 participants

Primary outcome timeframe

4 weeks after the last subject has completed the study

Results posted on

2024-12-19

Participant Flow

Participants randomized to each per sequence Arm are expected to remain in the same Arm throughout all intervention periods.

Participant milestones

Participant milestones
Measure
Cystagon First, Then Cystagon, Then RP103
Cystagon® dose in 50 mg and 150 mg capsule formulations administered every 6 hours in first intervention (after Run-in period) and RP103 dose in 25 mg and 75 mg capsule formulations administered every 12 hours in second intervention period.
Cystagon First, Then RP103, Then Cystagon
RP103 dose in 25 mg and 75 mg capsule formulations administered every 12 hours in first intervention (after Run-in period) and Cystagon® dose in 50 mg and 150 mg capsule formulations administered every 6 hours in second intervention period.
Run-in Period of 2-3 Weeks on Cystagon
STARTED
21
22
Run-in Period of 2-3 Weeks on Cystagon
COMPLETED
21
22
Run-in Period of 2-3 Weeks on Cystagon
NOT COMPLETED
0
0
First Intervention (3 Weeks)
STARTED
21
22
First Intervention (3 Weeks)
COMPLETED
21
20
First Intervention (3 Weeks)
NOT COMPLETED
0
2
Second Intervention (3 Weeks)
STARTED
21
20
Second Intervention (3 Weeks)
COMPLETED
21
20
Second Intervention (3 Weeks)
NOT COMPLETED
0
0

Reasons for withdrawal

Reasons for withdrawal
Measure
Cystagon First, Then Cystagon, Then RP103
Cystagon® dose in 50 mg and 150 mg capsule formulations administered every 6 hours in first intervention (after Run-in period) and RP103 dose in 25 mg and 75 mg capsule formulations administered every 12 hours in second intervention period.
Cystagon First, Then RP103, Then Cystagon
RP103 dose in 25 mg and 75 mg capsule formulations administered every 12 hours in first intervention (after Run-in period) and Cystagon® dose in 50 mg and 150 mg capsule formulations administered every 6 hours in second intervention period.
First Intervention (3 Weeks)
Withdrawal by Subject
0
1
First Intervention (3 Weeks)
Infection after pre-planned surgery
0
1

Baseline Characteristics

Phase 3 Study of Cysteamine Bitartrate Delayed-release (RP103) Compared to Cystagon® in Patients With Cystinosis

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
RP103 and Cystagon® Crossover
n=39 Participants
Per Protocol Population
Age, Categorical
<=18 years
36 Participants
n=5 Participants
Age, Categorical
Between 18 and 65 years
3 Participants
n=5 Participants
Age, Categorical
>=65 years
0 Participants
n=5 Participants
Age, Continuous
11.9 years
STANDARD_DEVIATION 4.33 • n=5 Participants
Sex: Female, Male
Female
16 Participants
n=5 Participants
Sex: Female, Male
Male
23 Participants
n=5 Participants
Region of Enrollment
France
13 participants
n=5 Participants
Region of Enrollment
United States
22 participants
n=5 Participants
Region of Enrollment
Netherlands
4 participants
n=5 Participants

PRIMARY outcome

Timeframe: 4 weeks after the last subject has completed the study

Outcome measures

Outcome measures
Measure
RP103
n=39 Participants
Per Protocol Population
Cystagon®
n=39 Participants
Per Protocol Population
The Steady-state White Blood Cell Cystine Levels of RP103 Compared to Cystagon®
0.5152 nmol ½ Cystine / mg protein
Standard Error 0.05555
0.4367 nmol ½ Cystine / mg protein
Standard Error 0.05555

SECONDARY outcome

Timeframe: 4 weeks after the last subject has completed the study

Outcome measures

Outcome measures
Measure
RP103
n=39 Participants
Per Protocol Population
Cystagon®
n=37 Participants
Per Protocol Population
Comparison of Cysteamine PK Profiles, Steady State Cmax, Between RP103 and Cystagon®.
2.73 Cmax (mg/L)
Standard Deviation 1.36
3.70 Cmax (mg/L)
Standard Deviation 1.72

SECONDARY outcome

Timeframe: 4 weeks after the last subject has completed the study

Outcome measures

Outcome measures
Measure
RP103
n=39 Participants
Per Protocol Population
Cystagon®
n=37 Participants
Per Protocol Population
Comparison of Cysteamine PK Profiles, Steady State Tmax, Between RP103 and Cystagon®.
72 Tmax (minute)
Standard Deviation 31
187 Tmax (minute)
Standard Deviation 89

SECONDARY outcome

Timeframe: 6 hours post dosing for Cystagon®; 12 hours post dosing for RP103.

Outcome measures

Outcome measures
Measure
RP103
n=39 Participants
Per Protocol Population
Cystagon®
n=37 Participants
Per Protocol Population
Comparison of Cysteamine PK Profiles, AUC(0-t), Between RP103 and Cystagon®.
357 AUC(0-t) (min*mg/L)
Standard Deviation 150
739 AUC(0-t) (min*mg/L)
Standard Deviation 334

Adverse Events

RP103

Serious events: 6 serious events
Other events: 16 other events
Deaths: 0 deaths

Cystagon®

Serious events: 1 serious events
Other events: 5 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
RP103
n=43 participants at risk
Safety Population during treatment periods
Cystagon®
n=41 participants at risk
Safety population during treatment periods
Musculoskeletal and connective tissue disorders
Knee deformity
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Metabolism and nutrition disorders
Hypovolaemia
0.00%
0/43 • Treatment periods
Safety population AE reporting
2.4%
1/41 • Number of events 1 • Treatment periods
Safety population AE reporting
Infections and infestations
Gastroenteritis
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Gastrointestinal disorders
Abdominal Discomfort
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Metabolism and nutrition disorders
Hypokalaemia
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Gastrointestinal disorders
Vomiting
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Injury, poisoning and procedural complications
Femur Fracture
2.3%
1/43 • Number of events 1 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting

Other adverse events

Other adverse events
Measure
RP103
n=43 participants at risk
Safety Population during treatment periods
Cystagon®
n=41 participants at risk
Safety population during treatment periods
Gastrointestinal disorders
Vomiting
11.6%
5/43 • Number of events 8 • Treatment periods
Safety population AE reporting
7.3%
3/41 • Number of events 5 • Treatment periods
Safety population AE reporting
Gastrointestinal disorders
Nausea
11.6%
5/43 • Number of events 7 • Treatment periods
Safety population AE reporting
4.9%
2/41 • Number of events 3 • Treatment periods
Safety population AE reporting
Gastrointestinal disorders
Abdominal Pain
7.0%
3/43 • Number of events 4 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting
Metabolism and nutrition disorders
Hypokalaemia
7.0%
3/43 • Number of events 3 • Treatment periods
Safety population AE reporting
0.00%
0/41 • Treatment periods
Safety population AE reporting

Additional Information

Evelyn Olson, Director

Horizon Pharma USA, Inc.

Phone: 224-383-3000

Results disclosure agreements

  • Principal investigator is a sponsor employee Investigators may publish or disclose study data, with the restriction that sponsor may embargo such communications for a period up to 60 days from the time submitted to sponsor. As a multi-center study, first publication of results will take place in conjunction with all investigators. If such a multi-center publication is not forthcoming within 18 months of study completion, individual investigators may publish as restricted according to limitations expressed above (60 day embargo).
  • Publication restrictions are in place

Restriction type: OTHER