Trial Outcomes & Findings for Phase II Confirmatory Study in Erythropoietic Protoporphyria (EPP) (NCT NCT01097044)

NCT ID: NCT01097044

Last Updated: 2019-10-28

Results Overview

The amount of direct sunlight exposure between 10:00 and 15:00 hours on days when no pain was experienced (e.g. 11-point Likert pain score of 0). Time was recorded in a patient dairy using 15 minute time blocks. The pain score is measured by the 11-point Likert Pain scale with minimum of 0 and maximum of 10. Likert Pain scale of 0 represents no pain and 10 represents worst imaginable pain.

Recruitment status

COMPLETED

Study phase

PHASE2

Target enrollment

77 participants

Primary outcome timeframe

Daily for 6 months

Results posted on

2019-10-28

Participant Flow

Study Recruitment period: April2010 - July2010 Study site location: Mt. Sinai, New York Carolinas Medical Center, North Carolina University of Alabama at Birmingham, Alabama University of Utah, Utah University of Texas Medical Branch, Texas University of California, San Francisco, California

Participant milestones

Participant milestones
Measure
Afamelanotide
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Overall Study
STARTED
39
38
Overall Study
COMPLETED
34
33
Overall Study
NOT COMPLETED
5
5

Reasons for withdrawal

Reasons for withdrawal
Measure
Afamelanotide
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Overall Study
Withdrawal by Subject
4
4
Overall Study
Lost to Follow-up
1
0
Overall Study
Withdrawal by Sponsor
0
1

Baseline Characteristics

Phase II Confirmatory Study in Erythropoietic Protoporphyria (EPP)

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Afamelanotide
n=39 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=38 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Total
n=77 Participants
Total of all reporting groups
Age, Continuous
38.1 years
STANDARD_DEVIATION 14.5 • n=93 Participants
42.6 years
STANDARD_DEVIATION 15.7 • n=4 Participants
40.3 years
STANDARD_DEVIATION 15.2 • n=27 Participants
Age, Categorical
<=18 years
3 Participants
n=93 Participants
0 Participants
n=4 Participants
3 Participants
n=27 Participants
Age, Categorical
Between 18 and 65 years
35 Participants
n=93 Participants
36 Participants
n=4 Participants
71 Participants
n=27 Participants
Age, Categorical
>=65 years
1 Participants
n=93 Participants
2 Participants
n=4 Participants
3 Participants
n=27 Participants
Sex: Female, Male
Female
16 Participants
n=93 Participants
18 Participants
n=4 Participants
34 Participants
n=27 Participants
Sex: Female, Male
Male
23 Participants
n=93 Participants
20 Participants
n=4 Participants
43 Participants
n=27 Participants
Region of Enrollment
United States
39 participants
n=93 Participants
38 participants
n=4 Participants
77 participants
n=27 Participants

PRIMARY outcome

Timeframe: Daily for 6 months

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

The amount of direct sunlight exposure between 10:00 and 15:00 hours on days when no pain was experienced (e.g. 11-point Likert pain score of 0). Time was recorded in a patient dairy using 15 minute time blocks. The pain score is measured by the 11-point Likert Pain scale with minimum of 0 and maximum of 10. Likert Pain scale of 0 represents no pain and 10 represents worst imaginable pain.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Time in Direct Sunlight Between 10:00-15:00 on Pain-free Days
8.25 Hours
Interval 0.0 to 48.3
0.75 Hours
Interval 0.0 to 71.3

SECONDARY outcome

Timeframe: Daily for 6 months

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

The days on which the participant experienced pain as a result of phototoxic reactions (caused by exposure to natural light) was recorded in a study diary. On each day such a reaction occurred, the participant scored the level of pain using an 11-point Likert pain scale, with minimum of 0 and maximum of 10. The 11-point Likert pain scale with a value of 0 represents no pain and 10 represents worst imaginable pain. The maximum severity of a phototoxic reaction was determined by the highest daily 11-point Likert scale score that occurred during that phototoxic reaction.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Maximum Severity of Phototoxic Reaction Experienced by Participants
5.0 score on a scale
Interval 0.0 to 8.0
5.0 score on a scale
Interval 0.0 to 10.0

SECONDARY outcome

Timeframe: Day 0, Day 60, Day 120, Day 180

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

Erythropoietic Protoporphyria Quality of Life Measure (EPP-QoL) is used to measure the quality of life of participants. The total EPP-QoL score ranges from 0 to 100, with a score of 0 as the worst quality of life and score of 100 as the best quality of life.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Quality of Life Measured by Participant Completed Questionnaire
Day 120
84.7 units on a scale
Interval 6.0 to 100.0
55.6 units on a scale
Interval 6.0 to 100.0
Quality of Life Measured by Participant Completed Questionnaire
Day 180
88.9 units on a scale
Interval 56.0 to 100.0
63.9 units on a scale
Interval 0.0 to 100.0
Quality of Life Measured by Participant Completed Questionnaire
Day 0
34.7 units on a scale
Interval 3.0 to 67.0
22.2 units on a scale
Interval 3.0 to 78.0
Quality of Life Measured by Participant Completed Questionnaire
Day 60
73.6 units on a scale
Interval 17.0 to 100.0
44.4 units on a scale
Interval 6.0 to 94.0

SECONDARY outcome

Timeframe: Baseline, Day 60, Day 120, Day 180

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

This was an exploratory assessment only to analyze whether afamelanotide-induced change in sun exposure would result in a reduction of protoporphyrin IX. The changes of the Total Protoporphyrin IX Level (μg/dL) from Screening Visit (ITT Population) were measured between the two groups. The Protoporphyrin IX level is a laboratory parameter that is measured in specialist labs.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Change of Total Protoporphyrin IX Level in Participants
Day 60
-471.3 mcg/dL
Standard Deviation 505.4
-628.5 mcg/dL
Standard Deviation 837.5
Change of Total Protoporphyrin IX Level in Participants
Day 120
-511.8 mcg/dL
Standard Deviation 595.3
-619.9 mcg/dL
Standard Deviation 1022.3
Change of Total Protoporphyrin IX Level in Participants
Day 180
-92.7 mcg/dL
Standard Deviation 549.1
-369.2 mcg/dL
Standard Deviation 1265.6

SECONDARY outcome

Timeframe: Daily for 6 months

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

The number of participants who experienced phototoxic reactions with Likert severity scores ≥ 4 and severity scores ≥7 were recorded. A derived endpoint was used. The number of participants who reported at least one phototoxic reaction with a Likert severity score of ≥ 4 was recorded. For severity scores ≥ 7, the number of patients who reported at least one phototoxic reaction with a Likert severity score of ≥ 7 was recorded. The 11-point Likert pain scale ranges from minimum of 0 to maximum of 10. The 11-point Likert pain scale with a value of 0 represents no pain and 10 represents worst imaginable pain.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Number of Participants With Phototoxic Reactions With Likert Severity Scores ≥ 4 and ≥ 7
Severity Likert score of ≥ 4
26 Count of Participants
24 Count of Participants
Number of Participants With Phototoxic Reactions With Likert Severity Scores ≥ 4 and ≥ 7
Severity Likert score of ≥ 7
7 Count of Participants
14 Count of Participants

SECONDARY outcome

Timeframe: Daily for 6 months

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

The days on which the participant experienced pain as a result of phototoxic reactions (caused by exposure to natural light) was recorded in a study diary. On each day such a reaction occurred, the participant scored the level of pain using an 11-point Likert pain scale, with minimum of 0 and maximum of 10. The 11-point Likert pain scale with a value of 0 represents no pain and 10 represents worst imaginable pain. The number of phototoxic reactions was determined by counting the number of episodes on which participants report a 11-point Likert scale score of 4 or more for one or more consecutive days.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Number of Phototoxic Reactions Experienced by Participants
2.0 Episodes
Interval 0.0 to 25.0
1.0 Episodes
Interval 0.0 to 13.0

SECONDARY outcome

Timeframe: Daily for 6 months.

Population: One active participant and one placebo participant withdrew after Baseline assessment and no data was provided for the analysis of this endpoint. The overall number of participants analyzed differs from the total number of study participants because the data was either incomplete and/or missing.

The days on which the participant experienced pain as a result of phototoxic reactions (caused by exposure to natural light) was recorded in a study diary. On each day such a reaction occurred, the participant scored the level of pain using an 11-point Likert pain scale, with minimum of 0 and maximum of 10. The 11-point Likert Pain scale with a value of 0 represents no pain and 10 represents worst imaginable pain. The total severity of phototoxic reactions was determined by the sum of daily 11-point Likert scale scores that occurred during phototoxic reactions. The overall sum of the severity per participant over the entire study was analyzed. The theoretical minimum score is 0 and the maximum possible score is 1800.

Outcome measures

Outcome measures
Measure
Afamelanotide
n=38 Participants
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=37 Participants
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Total Severity of Phototoxic Reactions Experienced by Participants Over the Entire Study
27.9 units on a scale
Standard Deviation 43.1
37.2 units on a scale
Standard Deviation 102.6

Adverse Events

Afamelanotide

Serious events: 2 serious events
Other events: 31 other events
Deaths: 0 deaths

Placebo

Serious events: 2 serious events
Other events: 29 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
Afamelanotide
n=39 participants at risk
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=38 participants at risk
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Gastrointestinal disorders
CRAMPY ABDOMINAL PAIN
2.6%
1/39 • 6 months (180 days)
0.00%
0/38 • 6 months (180 days)
Nervous system disorders
DIZZINESS
0.00%
0/39 • 6 months (180 days)
2.6%
1/38 • 6 months (180 days)
Cardiac disorders
Low Pulse Rate
0.00%
0/39 • 6 months (180 days)
2.6%
1/38 • 6 months (180 days)
Gastrointestinal disorders
RECURRENT RECTAL BLEEDING
2.6%
1/39 • 6 months (180 days)
0.00%
0/38 • 6 months (180 days)

Other adverse events

Other adverse events
Measure
Afamelanotide
n=39 participants at risk
Afamelanotide: One 16mg subcutaneous implant every 2 months for 6 months. (3 implants in total)
Placebo
n=38 participants at risk
Placebo: One placebo subcutaneous implant every 2 months for 6 months. (3 implants in total)
Gastrointestinal disorders
Nausea
20.5%
8/39 • 6 months (180 days)
5.3%
2/38 • 6 months (180 days)
Gastrointestinal disorders
Abdominal discomfort
7.7%
3/39 • 6 months (180 days)
5.3%
2/38 • 6 months (180 days)
General disorders
Fatigue
7.7%
3/39 • 6 months (180 days)
2.6%
1/38 • 6 months (180 days)
General disorders
Implant Site Pain
2.6%
1/39 • 6 months (180 days)
13.2%
5/38 • 6 months (180 days)
General disorders
Pyrexia
7.7%
3/39 • 6 months (180 days)
2.6%
1/38 • 6 months (180 days)
Infections and infestations
Nasopharyngitis
17.9%
7/39 • 6 months (180 days)
5.3%
2/38 • 6 months (180 days)
Infections and infestations
Sinusitis
2.6%
1/39 • 6 months (180 days)
7.9%
3/38 • 6 months (180 days)
Musculoskeletal and connective tissue disorders
Arthralgia
7.7%
3/39 • 6 months (180 days)
10.5%
4/38 • 6 months (180 days)
Musculoskeletal and connective tissue disorders
Back pain
7.7%
3/39 • 6 months (180 days)
13.2%
5/38 • 6 months (180 days)
Nervous system disorders
Headache
12.8%
5/39 • 6 months (180 days)
26.3%
10/38 • 6 months (180 days)

Additional Information

Dr. Dennis Wright, Acting Chief Scientific Officer

Clinuvel Pharmaceuticals Limited

Phone: 646 527 7310

Results disclosure agreements

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